A Microfabricated 3D Tissue Engineered "Liver on a Chip": High Information Content Assays for in vitro Drug Metabolism Studies by Anand Sivaraman Bachelor of Technology (Hons.), Chemical Engineering Indian Institute of Technology Kharagpur, India, 1999 Master of Science in Chemical Engineering Practice MassachusettsInstitute of Technology Cambridge, MA, June, 2002 Doctor of Philosophy in Chemical Engineering MassachusettsInstitute of Technology Cambridge, MA, August, 2004 © 2004 Massachusetts Institute of Technology All rights reserved I .I Signature of Author: .n Departrr) Certified by: - a,, 1 ef-rhical Engineering, August, 2004 -44 0//Dr.Linda %7 G.Griffith, s~jWor, Vq' Biological Engineering Division and Department of Mechanical Engineering Thesis Supervisor, August 2004 Accepted by: Dr.Daniel Blankschtein, Professor, Department of Chemical Engineering Chairman, Committee for Graduate Students, August 2004 MASSACHUS.iTS ' NS OFTECHNOLOGY ARCHIVES SEP0 2 2004 LIBRARIES A Microfabricated 3D Tissue Engineered "Liver on a Chip": High Information Content Assays for in vitro Drug Metabolism Studies by Anand Sivaraman Submitted to the Department of Chemical Engineering on August 9th2004, in partial fulfillment of the requirements for the degree of Doctor of Philosophy in Chemical Engineering Technical Summary Recent reports indicate that it takes nearly $800 million dollars and 10-15 years of development time to bring a drug to market. The pre-clinical stage of the drug development process includes a panel of screens with in itromodels followed by comprehensive studies in animals to make quantitative and qualitative predictions of the main pharmacodynamic, pharmacokinetic, and toxicological properties of the candidate drug. Nearly 90% of the lead candidates identified by current in vitro screens fail to become drugs. Among lead compounds that progress to Phase I clinical trials, more than 50% fail due to unforeseen human liver toxicity and bioavailability issues. Clearly, better methods are needed to predict human responses to drugs. The liver is the most important site of drug metabolism and a variety of ex vivoand in vitromodel systems have therefore been developed to mimic key aspects of the in vivo biotransformation pathways of human liver - a pre-requisite for a good, predictive pharmacologically relevant screen. Drug metabolism or biotransformation in the liver involves a set of Phase I (or p450 mediated) and Phase II enzyme reactions that affect the overall therapeutic and toxic profile of a drug. The liver is also a key site of drug toxicity following biotransformation, a response that is desirable but difficult to mimic in vitro. A major barrier to predictive liver metabolism and toxicology is the rapid (hours) loss of liver-specific functions in isolated hepatocytes when maintained under standard in itrom cell culture condition. This loss of function may be especially important in predicting toxicology, where the time scale for toxic response may greatly exceed the time scale for loss of hepatocyte function in culture. Although a wide variety of approaches to improving hepatocyte function in culture have been described, not all of the important functions specifically the biotransformation functions of the liver - can as yet be replicated at desired in ivolevels, especially in culture formats amenable to routine use in drug development. nttt fTcnlg Mascuet Massachusetts Institute ofTechnology ioehooyPoesEgneigCne iotechnology Process Engineering Center The in vivo microenvironment of hepatocytes in the liver capillary bed includes signaling mechanisms mediated by cell-cell and cell-matrix interactions, soluble factors, and mechanical forces. This thesis focuses on the design, fabrication, modeling and characterization of a microfabricated bioreactor system that attempts to mimic the in vivo microenvironment by allowing for the three dimensional morphogenesis of liver tissue under continuous perfusion conditions. A key feature of the bioreactor that was designed is the distribution of cells into many tiny (-0.001 cm3) tissue units that are uniformly perfused with culture medium. The total mass of tissue in the system is readily adjusted for applications requiring only a few thousand cells to those requiring over a million cells by keeping the microenvironment the same and scaling the total number of tissue units in the reactor. Using a computational fluid dynamic model in ADINA® and a species conservation mass transfer model in FEMLAB® , the design of the bioreactor and the fluidic circuit was optimized to mimic physiological shear stress rates at the tissue fluid interface, while satisfying the tissue oxygen demand. Using a broad spectrum of gene expression, protein expression and biochemical activity metrics, the liver tissue phenotype maintained during culture in the 3D bioreactor was seen to be substantially closer to that of native liver than that of cells maintained in standard cultures. Global transcriptional profiling was first used to identify genes that are differentially expressed between 2D collagen sandwich cultures, a variety of standard static 3D cultures, and the 3D perfused bioreactor at multiple time points up to 20 days in culture, relative to the expression profiles seen in liver in viv. The biotransformation genes (P450 and Phase II) of the liver were seen to be maintained at close to in ivo levels in the 3D perfused bioreactor, while a significant downregulation of these genes was seen in the other standard 2D and 3D static cultures. In order to validate the microarray data, a specific time point (Day 7 post isolation) was chosen to compare the basal and drug-induced expression levels of several important CYP450 and Phase II metabolism genes more quantitatively via RT PCR and biochemical activity assays. Classes of genes that were found to be differentially expressed between the tissues formed in the standard 2D collagen sandwich cultures and 3D perfused bioreactor cultures were found to be preserved at near in ivo levels in the 3D bioreactor even after seven days of culture following cellisolation from in tviv rat liver. In addition, the rates of biochemical activity in seven day old bioreactor cultures of the proteins that the biotransformation genes code for, as measured by the hydroxylation of testosterone, were found to be similar to rates measured in vivoas well as in freshly isolated hepatocytes. On the other hand, in keeping with the significant downregulation of the same genes in 2D cultures, the biochemical activities of the proteins in 2D collagen sandwich cultures were also seen to be significantly downregulated relative to in viwo.The ability of the 2D collagen sandwich cultures to inducers - 3-Methyl Cholanthrene, respond to prototypical drugs that are known to act as in imvo Technology Massachusetts Institute Institute of ofTechnology -4-4- Biotechnology Process Engineering Center Biotechnology Process Engieeriing Center Pregnelone-16a Carbonitrile, and Clofibric Acid - was seen to be sub-physiological unlike that of the 3D bioreactor cultures. Semi-quantitative RT PCR assays were used to compare basal expression levels of key hepatic nuclear receptors and transcription factors that regulate the induction of the CYP450 genes and other broad programs of liver-specific genes. The better maintenance of the liver specific transcription factors and nuclear receptors that regulate broad programs of liver function, in the 3D Bioreactor, may explain the superior basal and functional phenotype seen in the 3D perfused bioreactor. Some genes were seen to be significantly down-regulated in both the 2D collagen sandwich as well as well as the 3D bioreactor cultures, and based on current data available in literature, it is hypothesized that these genes are likely regulated by soluble factors present in the media. This study has attempted to show the utility of three dimensional tissue constructs as a more predictive screen for drug metabolism and xenobiotic induction, by looking at a very broad set of fundamental tissue phenotypic as well as regulatory markers. The study has also identified key regulatory mechanisms biotransformation that may be responsible for better retention of hepatic xenobiotic phenotype in 3D perfused cultures over 2D static cultures. Thus, it may be concluded that some important liver-specific functions of hepatocytes can be maintained at nearphysiological levels in vitr when isolated cells are cultured in a format that fosters tissue-like structures to form and allows continuous perfusion and convective distribution of nutrients over length scales comparable to those in the liver capillary bed. Because the functional unit in the microreactor comprises -1000 cells, the system can be readily scaled to meet a variety of needs. This thesis thus provides a foundation for extension of this culture model to applications where near-physiological levels of liver-specific expression are needed in long-term culture, including assessment of acute and chronic liver toxicity arising from exposure to drugs or environmental agents; models of disease such as viral hepatitis infection and cancer metastasis Thesis Supervisor: Dr.Linda G.Griffith Title: Professor of Biological Engineering and Mechanical Engineering Technology Massachusetts N-lassachusettsInstitute Institute of o Technology -5-5- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Acknowledgements I am greatly indebted to my advisor, Linda Griffith, for having given me the complete freedom and independence to carry out my thesis work. The idea of designing an in vitro3D liver tissue screen for metabolism and toxicity as part of my thesis, was one that came out of a visit to Schering-Plough Pharmaceutical Labs in June 2000 - and Linda was instrumental in having me visit SP and talk to the researchers there on current unmet needs in the field. To Steve Tannenbaum, my thesis committee member, I owe my deepest gratitude for many insightful discussions on the chemistry of metabolism of xenobiotics. I also warmly thank Roger Kamm and Mohammad Kazempur-Mofrad, for invaluable help and support with the mathematical modeling of the tissue culture system. I am grateful to Klavs Jensen for serving on my thesis committee, and providing valuable feedback on the effects of mass transfer on the intrinsic rates of metabolism of drugs. Ron White from Schering-Plough provided the much needed framework that helped ensure that the thesis was of appropriate relevance to the pharmaceutical industry. I must acknowledge several people who helped provide the technical expertise and assistance for this work. Dan Bauer and Emily Larson, for figuring the science behind the voodoo primary hepatocyte isolation protocols, Katy Wack and Donna Stolz for those wonderful electron microscopy pictures, Karel Domansky for help with the fabrication and design of the silicon scaffolds and the bioreactor, and finally Mark Powers, Carolyn Baker and Dena Janigian for their patience in teaching a chemical engineer what sterile techniques in biological experiments were all about. I have had the opportunity to work with two remarkable undergraduate researchers in Maritza Rodriguez and Seth Townsend. To Maritza, I owe many thanks for putting in those long nights with me in assembling and seeding reactors. To Seth, I possibly owe a very important part of my thesis - his validation of the 18s gene to normalize gene expression data, laid the foundation for many of the comparative gene expression studies across different systems. My sincere thanks to Tomo, Brad and Rebecca, for leading the effort on the Affymetrix microarray experiments and letting me include my reactor experiments with Technology Massachusetts Institute Massachusetts Institute of ofTechnology -6- -6 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center theirs. I wish to thank all of the Griffith and Lauffenburger lab members, past and present, for making my 5-year sojourn a wonderful learning experience. In particular, I thank Albert, Joe M., Katry,Nate, Artemis, Brent, Corey, Ajit, Kathryn, Megan, Emily, Tomo, Brad, Shawdee, Alexandria, Henrik, Dan, Adam, Jane, Steve, Dena and Joe S. for their friendships and for the wonderful times spent in their company in the BPEC lab and student office. Thanks are especially due to Brent Schreiber for having worked with me on reviewing the literature for the section on Liver Structure and Function (Section 1.1) and The in vivo microenvrmnment of the liverrevisited(Section1.2) - sections that the reader will find are common to both his thesis as well as mine. Finally, special thanks to Kevin - for being the surrogate advisor, the friend, philosopher and guide. Without his guidance and help, this thesis would not be in its present form. I will eternally value his friendship and support. Anand Sivaraman, MIT, Camb idge, MA August2004 Technology Massachusetts MIassachusetts Institute Institute of ofTechnology -7-7- Biotechnology Process Engineering Center Biotechnology Process Enginering Center Infond memory of my mother ... the most wonderful human being I have known ... Massahusets Massachusetts Institute ofTechnology Istitte -8-Biotchnoogy o Tecnolog -8- roces EgneigCtr Biotechnology Process Engineering Center Table of Contents Abstract .................................................................................................................................................... 2 Technical Summary................................................................................................................................- 3 Acknowledgements ................................................................................................................................- 6 - List of Figures......................................................... - 12- List of Tables ......................................................... 18 - Chapter 1..........................................................19 Introduction, Background and Motivation ........................................ ..................19 1.1 The Liver: Structure and Function - an engineering analysis .......................................-20 1.2 The in tevomicroenvironment of the liver revisited ........................................................27 1.3 Metabolism of Xenobiotics in the Liver ......................................................... - 29 1.4 The CYP450 biotransformation enzymes ........................................ ..................32 1.5 The Phase II biotransformation enzymes ..................................36 - 1.6The Drug Development Process......................................................... - 39 - 1.7 Hepatic Tissue Engineering for drug development assays: current status .................- 43 1.8 Hypotheses and Specific Thesis Objectives ......................................................... - 49 Chapter 2......................................................... 51 Design and Fabrication of the 3D Microfabricated Liver Bioreactor and Fluidics ................- 51 2.1 Key facets of the in tvit microenvironment that need to be recapitulated in an in itro system .............................................................................................................................................- 51 - 2.2 Design principles: biophysics of tissue morphogenesis ................................................. - 53 2.3 Microscopic design parameters........................................................................................... 55 2.4 Photolithography: fabrication of the silicon scaffolds ....................................................57 2.5 Design and fabrication of the polycarbonate bioreactor housing ................................-59 2.6 Assembly of the bioreactor ........................................................ - 61 2.7 Design of fluidic system ....................................................................................................... - 62 2.8 Maintenance of crossflow in the bioreactor ........................................................ - 63 2.9 Summary of key results and conclusions ........................................ .................68 Chapter 3........................................................ 69 - Modeling the Fluid Dynamics and Mass Transfer Effects in the 3D Bioreactor ...................- 69 3.1 Computational fluid flow model in ADINA®......................................................... - 70 3.2 Velocity profiles and shear stress distributions ........................................ ................- 74 3.3 Mass transfer model in FEMLAB ........................................................ - 78 3.4 Tissue disposition of oxygen ............................................................................................... - 82 3.5 Measurement of the effect of crossflow ........................................ .................90 3.6 Summary of key results and conclusions ........................................ ................ - 93 Chapter 4........................................................ 95 Basic Characterization of Liver Tissue Phenotype in the Bioreactor . ..............................- 95 4.1 Isolation of primary rat hepatocytes .................................................................................. - 96 4.2 Formation of multicellular spheroidal aggregates............................................................97 4.3 Bioreactor assembly and cell-seeding.........................................................98 4.4 Evaluation of hepatic phenotype: albumin and urea secretion.................................- 100 - Massachusetts Institute of Technology -9 - Biotechnology Process Engineering Center 4.5 Ultrastructural evaluation of tissue phenotype ............................................................- 102 4.6 Maintenance of p450 isoforms in the 3D bioreactor culture ......................................-105 4.7 Qualitative spectroscopic measurement of p450-1A activity ......................................-107 4.8 Summary of key results and conclusions ............................................................. - 109 - Chapter 5............................................................ - 110 - Measurement of Xenobiotic Metabolism in 2D and 3D Cultures .......................................... - 110 5.1 Challenges in the measurement of intrinsic reaction rates in a 3D system...............- 110 5.2 Paralyne coating of the fluidic circuit.............................................................111 5.3 Minimizing the dead volume in the bioreactor and fluidics........................................-116 5.3.1. Reservoirless fluidic circuit .............................................................- 116 - 5.3.2. Small reservoir, small bore tubing fluidic circuit .......................................................117 5.4 Measurement of xenobiotic metabolism in 2D collagen sandwich (2DCSW) and 3D Bioreactor (3DB) cultures......................................................... - 119 5.5 Theoretical validation of the zeroeth order Michaelis-Menten Kinetics at high substrate concentrations ............................................................125 5.6 Summary of key results and conclusions ............................................................. - 127 - Chapter 6............................................................ - 129 - Comparing Basal Biotransformation Capacity of various in vitroSystems .............................-129 6.1 Global gene expression profiling of tissue phenotype ................................................. - 129 6.2 Quantifying relative gene expression: The two step process: Reverse Transcription Polymerase Chain Reaction (RT-PCR) ..........................................................134 6.3 Identification of a well-conserved housekeeping gene for RT-PCR studies............- 138 6.4 Analysis of RT-PCR data: Use of normalized fold change as a metric to compare gene expression data......................................................... - 141 6.5 Comparing liver specific gene expression between in vitri cultures using Affymetrix ® microarray and RT-PCR studies..........................................................145 6.6 Relative expression of liver transcription factors between 2D and 3D cultures .....- 150 6.7 Measurement of testosterone metabolism in 2DCSW and 3DB cultures ................- 151 6.8 Biochemical regulation of tissue function: effect of soluble factors in the media... - 154 6.9 Summary of key results and conclusions ............................................................. - 156 Chapter 7............................................................ - 158 Inducibility as a Quantitative Functional Response Marker .....................................................158 7.1 The cue-signal-response hypothesis.............................................................159 7.2 Inducibility of 2D and 3D cultures using 3MC and clofibric acid .............................-164 166 7.3 Inducibility of 2D and 3D cultures using PCN ............................................................7.4 Key regulatory mechanisms that affect p450 basal expression and induction .........- 169 7.5 Relative inducibility of 2D and 3D cultures - a summary ...........................................171 7.6 Summary of key results and conclusions ............................................................. - 173 C hapter 8............................................................................................................................................. - 175 Conclusions and Recommendations ...................................................175 References ..................................................- 180 - Appendices: Protocols and Experimental Methods ...................................................196 ® Isolation of primary rat hepatocyte enriched fraction using Percoll ..............................197 Isolation of total RNA from isolated hepatocytes, in vivo tissue, 2D and 3D cultures- 198 - Massachusetts Institute Massachusetts Institute of of Technology echnology -10-10- Biotechnology Process Engineering Center Biotechnology Pocess Engineering Center Primer design procedures and guidelines ........................................ ............... - 203 cDNA Preparation and Reverse Transcriptase-Polymerase Chain Reaction (RT-PCR) protocols ...................................................................................................................................... -205 Hepatocyte isolation and Spheroid Formation ........................................................209 Bioreactor Seeding and Maintenance of Culture........................................................ - 210 Measurement of the total number of viable cells in cultures using the measured amount of total RNA and RT-PCR against the 18s gene........................................................212 Preparation of 2D collagen gel sandwich cultures ........................................................ - 213 Preparation of 3D spheroid and 3D Matrigel spheroid cultures ......................................-214 Induction experiments on 2D collagen sandwich and 3D microreactor cultures .........- 215 Accession numbers for mRNA and complete cds sequences used .................................-216 Primer sequences used in RT-PCR studies ........................................ ................217 Melting and annealing temperatures of designed primers ..................................................219 Microarray Processing ...............................................................................................................221 Testosterone Metabolism studies ............................................................................................222 The Drug Development Process - a detailed overview .....................................................224 - Inttt ofTcnlg Mascuet Massachusetts Institute of Technology I-BoehooyPoesEgneigCne Biotechnology Process Engineering Center -11 - List of Figures Figure 1-1:The functions of the liver ...................................................- 21 - 23 Figure 1-2: The multiple models of liver architecture...................................................................Figure 1-3: Schematic drawing of the structure of the normal liver .........................................24 Figure 1-4: Schematic of the Liver Sinusoid...................................................- 25 - Figure 1-5: Comparative state of liver non-parenchymal cells in normal vs damaged states - 27 Figure 1-6: The role of p450's in medicine and biology...............................................................33 Figure 1-7: Catalytic cycle of CYP450's...........................................................................................34 Figure 1-8: Relative abundance of the various p450's in human liver .......................................-35 Figure 1-9: Co-factors involved in Phase II biotransformation reactions ................................-38 Figure 1-10:A schematic of the drug discovery and development process .............................-39 Figure 1-11: The main causes of failure of lead candidates ................................................... - 40 Figure 1-12: The growing expenditure on predictive in vitro and in vivmanimal models of toxicology icolo.41................................................................................................................................................- - Figure 1-13: In vitroand in vivomodels used in the development of new drugs, ranging from human to isolated enzymes, in order of in vivoresemblance ................................................... - 43 Figure 1-14:Advantages and disadvantages of the various in vitromodels ...............................-45 Figure 1-15:Advantages and disadvantages of the isolated hepatocytes and hepatocyte cultures as an in vitromodels ...................................................- 46 - Figure 1-16: Applicability of current in vitro models in various stages of biotransformation research ....................................................................................................................................................- 49 - Figure 2-1(a): Important facets of the in vivomicroenvironment that need to be recapitulated in an in vitrosystem....................................................- 52 - Figure 2-1(b): Important facets of the in vivo microenvironment that need to be recapitulated in an in vitrosystem....................................................- 53 - Figure 2-2: Cell-Cell homotypic interactions that lead to histotypic re-organization over a defined length and time scale and adhesion guided morphogenesis of pure and mixed cell 54 populations............................................................................................................................................- nsitteofTehnloy1-12- Masahuets Nassachusctts Institute of Technology Prcs nieeigCne Bitcnlg Biotechnology Process Eineering Center Figure 2-3: The design parameters used in the fabrication of the cell-holding channels in the silicon scaffold ......................................................................................................................................57 Figure 2-4: Schematic of the DRIE process (left) used to fabricate the scaffolds and photos of the channel structure and geometries showing resolution. .......................................................... - 58 Figure 2-5: Silicon scaffolds of different geometries microfabricated by DRIE .....................- 59 Figure 2-6: Changes made to the design of the MilliF reactor, based on feedback from experiments on MilliII reactors ............................................................- 60 - Figure 2-7: An expanded view of the scaffold assembly (left) and a schematic of the various parts of the bioreactor .......................................................... - 61 - Figure 2-8: Photograph of an assembled bioreactor ........................................................... - 62 - Figure 2-9: Single pump fluidic system used to run culture media through the bioreactor.. - 63 Figure 2-10: Loss in crossflow seen in 3D bioreactors 24-48 hrs. post seeding......................- 64 Figure 2-11: Schematic showing a two-pump fluidic system where the second crossflow pump pushes fluid from the top of the reactor in through the channels ...............................................64 Figure 2-12: Schematic of the two-pump fluidic circuit after the addition of the inline filters- 65 Figure 2-13: Cell debris seen in the media in the reservoir 1.5 hrs. after reversal of crossflow- 66 Figure 2-14: Cleaning effect of crossflow ........................................ ................... - 67 - Figure 2-15: Schematic of the optimized two-pump fluidics used with the bioreactor .........- 67 Figure 3-1: Scanning electron micrographs providing appropriate length scales for the idealized tissue geom etry .....................................................................................................................................72 Figure 3-2: Idealized tissue-channel geometry with a cylindrical conduit for convective flow- 73 Figure 3-3: Mesh distribution in the ADINA® model used to describe fluid flow in the bioreactor ............................................................- 73 - Figure 3-4: Uniform flow seen over the top of the channels, over most part of the bioreactor after a short entrance length................................................................................................................ 74 Figure 3-5: Uniform axial flow is .seen to be maintained over most of the channels .............- 75 Figure 3-6: Velocity profile in a single channel of the bioreactor .............................................. - 76 Figure 3-7: Shear stress distribution along the depth of a channel ............................................76 - Technology Massachusetts MlassachusettsInstitute Institute of ofTechnology 13 - 13- Biotechnology Process Engineering Center Biotechnology Pcess Engineering Center Figure 3-8: Uniform shear stress distribution seen over the top of the channels in the 77 - bioreactor ..............................................................- Figure 3-9: Maximum shear experienced by the cells in a channel as a function of crossflow rate ..............................................................- 78 - - 80 Figure 3-10: Interfaces and boundaries in the mass transfer model .......................................... Figure 3-11: Appropriate non-dimensional parameters used to characterize the system.......- 81 Figure 3-12. Tissue distribution of oxygen in the limiting case of reactors with zero cross-flow - Scenario 1 ..............................................................- 85 - Figure 3-13: Tissue distribution of oxygen in bioreactors with cross-flow - Scenario 1......- 86 Figure 3-14. Tissue distribution of oxygen in the limiting case of reactors with zero cross-flow: Scenario 2:.............................................................-.................................................................................. 88 - Figure 3-15: Tissue distribution of oxygen in bioreactors with cross-flow - Scenario 2 .......- 89 Figure 3-16: Minimum tissue oxygen concentration asymptotically falls to zero for large values of the Damkohler number (Da) ........................................................................................................90 Figure 3-17: Effect of perfusion crossflow rates on tissue expression of CYP3A2 ...............-91 Figure 3-18: Expression of Hif-3-ocmRNA in bioreactor cultures operated at different crossflow rates using RT-PCR ............................................................. - 92 - Figure 3-19: Induction of Heme Oxygenase - 1 mRNA in bioreactor culture under hypoxia:- 93 Figure 4-1: Schematic of the steps involved between the isolation and seeding of rat liver cells into the bioreactor ............................................................. 95 - - Figure 4-2 A schematic of enriched rat liver hepatocyte isolation procedure ..........................-96 Figure 4-3: Schematic diagram of the bioreactor fluidic system. ...................................- 98 - 100 Figure 4-4: Cleaning effect of crossflow .......................................................................................101 Figure 4-5: Albumin secretion rates in bioreactor cultures ......................................................102 Figure 4-6: Urea secretion rates in 3D bioreactor cultures........................................................103 Figure 4-7: TEM's of tissue phenotype in the bioreactor .........................................................Figure 4-8: SEM's of tissue structures formed in the micro channels of the bioreactor .....- 104 Figure 4-9: Toluidine blue stained sections of the liver tissue from a bioreactor channel..- 105 - Inttt fTcnlg Massachusetts Massachusetts Institute ofTechnology 1-BoehooyPoesEgneigCne -14Biotechnology Process Engineering Center Figure 4-10: Maintenance of expression of CYP450 2B protein for at least upto two weeks in bioreactor culture ............................................................- 106 - Figure 4-11: Repeatability of p45 0 expression in bioreactor cultures ......................................-107 Figure: 4-12: Spectrometric system used for in situ spectroscopy of the 3D perfused tissue- 107 Figure: 4-13: The EROD Assay to measure the activity of P4501A .......................................-108 Figure 4-14: Detection of CYP activity using a fluorescence detector .................................- 109 Figure 5-1: Loss of hydrophobic drug to adsorption, absorption and non-specific binding in empty reactors with no cells seeded............................................................- 112 - Figure 5-2: Parylene N was used to passivate the pump tubings ............................................. - 113 Figure 5-3: Steps in the conformal deposition of Parylene N on CFLEX® tubing .............- 114 Figure 5-4: Minimal loss of hydrophobic drug to Parylene N coated tubing ........................-115 Figure 5-5: Minimal loss of hydrophilic products of metabolism due to ...............................-116 Parylene N coated tubing ...................................................................................................................116 Figure 5-6: Reservoir-less reactor fluidics configuration to minimize dead volume ............- 117 Figure 5-7: Components of the two pump, small reservoir fluidic circuit .............................-118 Figure 5-8: Testosterone biotransformation pathway in the rat liver....................................- 119 Figure 5-9: Steps in quantifying the concentration of hydroxylated products of testosterone metabolism using HPLC-UV ...........................................................- 121 - Figure 5-10: Specimen standard curve used to calculate the normalized slope of AUC ratio- 124 Figure 5-11: Specimen absorbance spectrum of a hydroxylated product and recorded retention time .. .........................................................-............................................................................................. 12 5 Figure 5-12: A schematic analysis of the limiting case of high concentration of drug (substrate) added to the 3D culture ............................................................- 126 - Figure 5-13: Results for the tissue distribution of testosterone ................................................127 Figure 6-1: Stable expression (though downregulated in many cases) of many of the CYP genes is seen in the 2D collagen sandwich cultures after seven days in culture .................................-132 Figure 6-2: Log ratios of all culture systems (2D, 3D) versus intact liver tissue were clustered for all CYP450 genes as represented on the Affymetrix Rat Genome (U73A) arrays...........- 133 - 1n5-t fTcnooy-1-Boecnlg ~~~~~ Massachusetts~~~~~~~ I2assachusettsInstitute ofTechnology -15- rcsEgneigCne Biotechnology Process Engineering Center Figure 6-3: The various steps involved in an RT-PCR experiment used to quantify relative 134 gene expression....................................................................................................................................Figure 6-4: The three regions seen in the fluorescence-cycle number amplification curve obtained following RT-PCR ..........................................................- 135 - Figure 6-5: Fluorescence following binding of the SYBR Green dye to ds-DNA ...............- 135 Figure 6-6: Linear relationship between threshold cycle number and quantity of gene product for the 18s gene ...................................................................................................................................137 Figure 6-7: Isolated Hepatocytes vs. Total RNA ..........................................................- 139 - Figure 6-8: 18s gene per cell is invariant across the different culture systems.......................-140 Figure 6-9: Effect of addition of testosterone on the per cell 18s expression levels in 2D collagen sandwich cultures ..........................................................- 141 - Figure 6-10: 18s versus b-actin as a normalization gene .........................................................- 145 Figure 6-11: Relative CYP450 gene expression across cultures - from RatU34A microarray- 147 Figure 6-13: Basal expression of hepatic transcription factors in isolated hepatocytes, 2D collagen sandwich culture (day 7), and 3D microreactor (day 7) cultures expressed as log2-fold change relative to liver in vivo.............................................................................................................151 Figure 6-14: Downregulation of Bile and Fatty Acid transporters seen in both 2D as well as in 3D reactors ..........................................................................................................................................155 Figure 6-15: Other genes modulated by specific biochemical components in the media ... - 156 Figure 7-1 : Prototypical inducers and nuclear receptors that mediate the induction of some of the important CYP450's found in rat liver.....................................................................................162 Figure 7-2: Role of nuclear receptors in CYP gene induction. .................................................163 Figure 7-3: Dose response in 2D collagen sandwich cultures: Case of PCN ........................-163 Figure 7-4: Mechanism of induction of 3MC-responsive genes .............................................. - 164 Figure 7-5: Mechanism of induction of Clofibric Acid-responsive genes ..............................-165 Figure 7-6: Results of prototypical induction studies: 3MC, CLO studies .............................-166 Figure 7-7: Mechanism of induction of PCN responsive genes ....................................- 167 - Figure 7-8: Results from the PCN Induction studies .......................................................... - 168 Figure 7-9: Basal expression of ligand-binding nuclear receptors ............................................170 - Inttt fTcnlg Massachusett Massachusetts Institute ofTechnology 1- -16- Prcs nieeigCne Bitcnlg Biotechnology Process Engineeting Center . Figure 8-1: A one-pass back mix fluidic design for the measurement of one-pass extraction rates at non-saturable concentrations of the drug....................................- Massachusetts Massachusetts Institute Institute of of Technology Technology 17-17- 176 - Biotechnology Process Engineering Center Biotechnology Process Engineeing Center List of Tables Table 1-1: General pathways of xenobiotic biotransformation and their...................................- 31 31 major sub-cellular location.......................................................................................................................Table 3-1. Average viable cell numbers and non-dimensional parameters for modeling the tissue distribution of oxygen in bioreactors with and without cross-flow - Scenario 1 .........................-84 Table 3-2. Average viable cell numbers and non-dimensional parameters for modeling the tissue distribution of oxygen in bioreactors with and without cross-flow - Scenario 2 .........................-87 Table 5-1: Products of testosterone hydroxylation and p450's that mediate their formation. - 123 Table 6-1: Global comparison of gene expression profiles across different in vitrocultures at various tim e points..................................................................................................................................- 131 - Table 6-2: Higher basal activity of 2C11, 2B1, 2B2 and 3A1, 3A2 seen in the 3D microreactors 153 over the 2D collagen sandwich cultures .............................................................................................- Masahset Massachusetts Institute of Technology Isitt o -18- ecnloy-1-BoehogyPcssngeengCte Biotechnology Process Engineering Center Chapter 1 Introduction, Background and Motivation Recent reports indicate that it takes nearly $800 million dollars and 10-15 years of development time to bring a drug to market [1]. In itromodels are used to make quantitative and qualitative predictions of the main pharmacodynamic, pharmacokinetic, and toxicological properties of the candidate drug, during the pre-clinical screening stage of the drug development process [2-4]. Nearly 90% of the lead candidates identified by current in vitro screens, fail to become drugs [5], resulting in an unmet need for systems that are more predictive of the in vivo metabolism and toxicity profile of the drug. A more predictive in vitro system can help fail drugs earlier in the drug development process, much before expensive clinical trials. Drug biotransformation that involves a set of Phase I (or p450 mediated) and Phase II enzyme reactions [6, 7], can affect the overall therapeutic and toxic profile of a drug. It occurs in many tissues, with the liver as the most important organ [7]. Thus, a variety of in ditr model systems have been developed to mimic the in vivo biotransformation pathway in the liver - a pre-requisite for a good, predictive screen. These include isolated perfused livers, liver tissue slices, primary cell culture and suspension culture systems, isolated organelles, membranes or enzymes, and a variety of recombinant systems [2-4, 8]. Each model has its advantages and disadvantages, and these have been well documented in literature [2-4, 8-10]. One such in itromodel, the primary isolated hepatocytes, rapidly lose liver specific functions when maintained under standard in vitro cell culture conditions [8, 11, 12]. A variety of modifications to conventional culture methods have been developed to foster retention of hepatocyte function, including culture on or in basement membrane gels [8, 13], co-cultures with other liver-derived or non-liver cell types [14-17], cultures in collagen gel sandwiches [18], three dimensional culture in spheroids [19, 20], roller bottles [21, 22], addition of exogenous compounds to the culture medium [23], culture of cells in a variety of bioreactors [24-27]. Still, not all of the important functions -specifically the biotransformation functions of the liver can Technology Massachusetts Mc~assachusetsInstitute Institute of ofTechnology - 19 19- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center as yet be replicated at desired levels, prompting continued development of new culture methods. Dr. Jim Darnell who won the Lasker award in the year 2002, discovered that "a liver cell could remain a liver cell only when it stayed in the liver .... without constant signals from their normal place in the organ, liver cells lost a crucial molecular component ... that helps give them their identity" [28]. Thus, in essence, it is important that all the important facets of the in vivomicroenvironment be recreated in any in tr liver tissue culture system to replicate all the higher order functions of the organ system. A broad goal of this thesis is to recreate the in vivo3D microenvironment that enables cell-cell, cell-matrix, soluble and shear mediated signalling mechanisms in an in vitrosystem, in an attempt to better preserve the biotransformation functions of the liver in long term cultures. Such an in vitro system may help address a number of unmet needs in the pharmaceutical industry. It may find useful applications as a screen for quantitative studies on the metabolism and toxicity of new chemical entities (NCE's), as well as a model for studying the pathology of chronic liver diseases and infection by Hepatitis C [29] This chapter starts with an introduction to the basic biology of the liver, followed by a primer on the role of the liver as an organ in the biotransformation of xenobiotics - Le. the ability to clear 'foreign' substances from the body. Following a discussion on current challenges that face the Pharmaceutical industry - specifically the the drug development process, the role of hepatic tissue engineering in developing screens for metabolism and toxicity is briefly discussed. This chapter concludes with a note on the central hypothesis on which this thesis is based followed by a discussion on specific thesis objectives. 1.1 The Liver: Structure and Function - an engineering analysis The liver possesses an extremely sophisticated engineering design. It houses a large, highly structured reactor bed, an intricate flow manifold, and a separation system that efficiently delivers metabolic products to the blood stream, while shunting bile salts into the retrograde bile duct. This system's main functions are to remove toxins and provide metabolic Masachsets echoloy Istiuteof -0 -20- MlassachusettsInstitute ofTechnology -Bioecholoy EgeeigCte Poces Biotechnology Process Engineering Center activity such as cytochrome P450 activity, glycogen storage, urea production, and release of proteins, carbohydrates, lipids and cholesterol, and metabolic wastes. In addition the liver serves as a storage vessel for iron (processed from hemoglobin) and copper, fat-soluble vitamins (A, E, D, and K), and blood, which can be released during hemorrhage. In all, over 500 functions have been identified in the liver many of which are vital to sustain life. Some of these functions are schematically depicted in Figure 1-1. These disparate functions arise from the highly intricate cellular arrangement and structure. 11a"W; /1{ , '"I Figure 1-1: The functions of the liver There are two main competing views of the structural organization of the fundamental functional unit of the liver - the lobule and the acinus [30]. Both models posses a hexagonal tissue structure with the portal triads at the vertices and the central vein at the centroid. In IKieman's proposed lobule model [31] the blood passes into the periphery from the digestive tract via the portal triad, traverses the sinusoid, and then exits via the central vein (hepatic vein) (Figure 1-2A, [32]. The portal triad is comprised of three vessels: the hepatic artery bringing Massachusetts Miassachusetts Institute nstitute ofTechnologv of Technology -21-21 - Biotechnology Process Engineering Center BiotechnolojW Process Engineeringr Center oxygenated blood from the heart, the portal vein carrying enriched blood from the intestine, and the bile duct which drains bile from individual bile ducts (Figure 1-2B). These inputs and outputs branch into complex tree structures, which supply and drain the entire liver. Rappaport proposed the acinar model in 1954 based on the observation that as blood passes through the sinusoids, oxygen content, and dissolved solutes are altered at different positions in the sinusoid by the hepatocytes which have contacted it. Consequently, the cell types in the liver represent a heterogeneous population of cells whose function differs relative to the composition of contacted blood. Therefore, the acinus is subdivided into three zones graded by the depletion of oxygen and other metabolites in adjacent RBC's as they travel the length of the sinusoid toward the central vein (Figure 1-2C) [33]. Isolating a single sinusoid shows the fundamental unit of liver structure: a set of thin hepatocyte plates, called the acinus, strung between the portal triad and the hepatic veinule (Figure 1-2D) [34-36]. These two models of the liver, though seemingly disparate, provide the foundation for current models. Further research should result in a more definitive model for the architecture of the basic functional unit of the liver, but for the purposes of this research the liver will be viewed as a heterogeneous hexagonal tissue, which is a simplification and an incorporation of both theories. Additionally the liver has an equally intricate organization on the cellular level that may be required to be replicated to achieve proper function. The acinus is organized in a perfused, spongelike, capillary bed structure, composed primarily of mature hepatocyte plates of a single cell thickness, known as the parenchyma [30]. These plates have an apical domain which forms bile canalicular networks involved in the secretion of bile components and metabolites of xenobiotics, and a basal domain which interacts with ECM and participates in cell signaling. These hepatic plates are lined by fenestrated endothelial cells, which create a physical and chemical shield between the sinusoid and the hepatic plate. The region between the endothelium and the hepatic plate, known as the Space of Disse, is traversed by Stellate cells, the resident liver fibroblasts. Interspersed in the sinusoid are kupffer cells, a specialized form of macrophage (Figure 1-3A). Fluid flows through two paths: the bulk travels "down" the acinus from the portal region to the central vein; hepatocytes also form ducts known as bile Massachusetts Institute of Technology - 22- Biotechnology Process Engineering Center canaliculi that transport bile retrograde or "up" to the bile duct in the portal triad (Figure 13B). These ducts are separated from the rest of the tissue by tight junctions between neighboring hepatocytes in a similar fashion to that of the digestive system [34-36]. A. B. Wnr C. D. Figure 1-2: The multiple models of liver architecture (A) Liver microarchitecture features hepatocytes around the central vein (B) Liver lobule showing the portal triad, hepatic sinusoids and blood flow regions (C) Regions of the classical lobule and acinus. (D) Schematic of liver operational units: Combination of the classical lobule and the acinus (ImageA isfr m Jaregui et al. 2000, B and Cjfim Klassenet al. 2001, and D fomJunguiera et al. 1999) Technology Institule of Massachusetts ;Massachusetts Institule ofTechnology 23 -23 - Biotechnology Process Engineering Center Biotechnoogy Process Engineering Center A. B. tery Centralvein- -Liverplates Kupffer cells- Bile canaliculus Endothelialcellsof sinuso -Fat-storing cell -Sinusoidal capillary -Fat-storing cell -Hering'scanal in Inletarteriole _Inletvenule Inletvenule- -Distributing vein Hepaticartery Portalvein- Bile duct Distributing ve Figure 1-3: Schematic drawing of the structure of the normal liver. (A) Sinusoidal flow conduits and cellular positioning (B) Vascularization of one portal triad unit Images Taken from Junqueira and Carneiro,Basic Histology, a text and atlas,p. 333, Figure 16-1 1. The normal hepatic sinusoid is formed or lined by four cell types, each with its specific phenotypic characteristics, functions, and topography (Figure 1-4). These cells may be considered to represent a functional unit at the border between the hepatic plates and the blood. They participate in various liver functions and liver pathologies and our knowledge about this contribution is growing (emerging as a new focus in liver research). The heterogeneity of these cell-types and possible cooperation between NPC's and the hepatocytes may add to the overall understanding of liver function. Massachusetts MlassachusettsInstitute nstitute of of Technology Technolop, - 24 24- - Biotechnology Process Engineering Center Biotechnolop, Process Engineering Center A. B. ~ ~ j I _ .. 1: _r- ,,, $1museid -1-* __ i0 _ _ , . , ~~ _.~' an~ 1~i c EDO~ Figure 1-4: Schematic of the Liver Sinusoid (A) Location of NPC's in reference to a capillary of the sinusoid (B) Detailed architecture of sinusoidal microarchitecture (Image (A) isfrom the thesis of Tomo Iida, 2003, (B)from Wake et al. 1997) This section briefly reviews the main function and phenotypic characteristics of different cell types that constitute a sinusoid: Hepatocytes Hepatocytes are highly differentiated epithelial cells that form the cell plates of the liver lobule and perform the majority of the previously described functions attributed to the liver. In vivo liver is composed primarily of hepatocytes (-60-65%) [30], that function in detoxification of systemic and portal blood; secretion of plasma proteins, growth factors, and bile; metabolism of proteins, steroids, and fat; and storage of vitamins, iron, and glycogen [4, 8]. Endothelial Cells Endothelial cells (EC's) constitute the closed lining or wall of the capillary (Figure 1-3) and make up 18-2 3 % of all hepatic cells. EC's posses small fenestrations to allow the free diffusion of substances, like 02, but not of particles like chylomicrons, between the blood and the hepatocyte surface. This filtering effect regulates the fat uptake by the liver. These cells Massachusetts Massachusetts Institute Institute of of Technology Technology -25 -25 -- Biotechnology Process Engineering Center Biotechnology Process Engineering Center also have a pronounced endocytotic capacity, which makes them an important part of the reticuloendothelial system [37]. They are also active in the secretion of bioactive factors and extracellular matrix components of the liver. Zonal heterogeneity of the endothelial lining has recently been reported with regard to its filtering capacity (fenstration) and binding capacity for lectins and cells [38]. Kupffer Cells Kupffer cells (KC's) line the sinusoids of the liver and are attached to the endothelial cells (Figure 1-4) and represent 8-12% of hepatic cells. They are derived from blood monocytes and are the largest group of fixed cells macrophages in the body [30]. They are potent mediators of the inflammatory response by the secretion of a variety of bioactive factors and play an important role in the immune defense. KC's are have a high endocytotic capacity and are capable of removing particulate matter from the bloodstream. They phagocytose old cells, foreign particles, tumor cells, bacteria, yeast, viruses, and parasites (Valatas et al. 2003). The large sizes of the liver and tremendous numbers of kupffer cells make the sinusoids a very important location for clearance of particulate matter from the plasma. About one-third of the hearts cardiac output flows through the liver and makes it a key source of plasma filtration. Kupffer cells are known to be numerically more prominent in the periportal region [39]. Stellate Cells The stellate cells (also called Ito cells or lipocytes) lie in the space of Disse, encircling the sinusoidal endothelium and represent 5-8% of hepatic cells [40] . These cells are the main hepatic source of extracellular matrix components and are thought to be the body's main site of Vitamin-A storage [30]. These cells are the only sinusoidal phenotype capable of forming junctions with hepatocytes [41]. Stellate cells display two phenotypes in normal in vimotissue. In the resting or 'quiescent' state, they resemble fibroblasts but their cytoplasm contains numerous droplets in which Vitamin-A is stored. Upon transdifferentiation to the 'activated' state stellate cells elongate to resemble myocytes (Figure 1-5) and exhibit contractile function that plays a role in regulation of sinusoidal tone and resistance. In chronic liver disease the stellate cells synthesize and secrete collagen into the Space of Disse, leading to "capillarization" Technology Massachusetts Institute Massachusett Institute of ofTechnology - 26 26- - Biotechnology Process Engineering Center Biotechnology Process Engineeringg Center _____ (fibrosis) of the sinusoids (Figure 1-5). Stellate cells are more numerous in the periportal region than in the pericentral region of the hepatic acinus. Periportal cells also store higher amounts of Vitamin-A. * A NT-'.rml It;.S.. ... HepattLlc PralI B rIR~g I NcPll? 4yofibroblas I~irit fal/mgbra PortalArhnOZ ulrTr Cl.l nil.. ijm / .iatfix 'lknpavirkma wr/ llPiltf~r Prokriting Myribroblast t Im WMrrbraRd Far Figure 1-5: Comparative state of liver non-parenchymal cells in normal vs damaged states Pit Cells These are known to represent a liver-associated population of large granular lymphocytes. They have the capacity to kill tumor cells and probably also play a role in the antiviral defense of the liver. The have been suggested to have a growth-regulatory function in the liver [30]. Like the kupffer cells, they are known to be more abundant in the periportal region of the sinusoid [42, 43]. 1.2 The in vivomicroenvironment of the liver revisited A major challenge in toxicology and pharmacology research has been to create an in vivo like primary cell culture analog that maintains liver-specific function and replicates biological tissue features such as polarity, architecture, and normal bile canaliculi formation, while maintaining the biotransformation capacity [8, 12]. A variety of culture systems have demonstrated the retention of original morphological characteristics and the maintenance of some hepatic function [8, 12, 14, 16, 18, 24, 27, 44, 45], however, none of these systems have succeeded in replicating the liver associated environmental cues fully and therefore have not been successful in stabilizing the hepatic phenotype [24, 45-47]. Technology Massachusetts Massachusetts Institute Institute of ofTechnology 27 -27 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Sinusoidal cells change phenotype drastically depending on their environment. This 'transdifferentiation' is pronounced enough that it is not clear whether the gene expression differences between cell types or differences between phenotypic states of one of the cell types have a greater dynamic range [48, 49]. Thus, even though a particular sinusoidal cell type has a restricted set of expressible genes, the specific proteins that are expressed within that subset can change drastically in response to a given environment. Therefore, it is important to replicate the sinusoid microenvironmental cues within an in vitroculture system. 1) Cell-MatrixInteractons Hepatic stability and polarity have been shown to be influenced by variations in composition and topology of extracellular matrix (ECM) [8, 12, 38, 50]. ECM interacts with cells via binding of intergrins, and other cell surface adhesion receptors and activate a number of intercellular signaling mechanisms, which enhance homeostasis of cell phenotype [51]. Culture of cells with collagen type I or Matrigel® (biologically derived, basal lamina like compound) have induced cells to maintain phenotype specific morphology and express liverspecific function longer than previous hepatocyte monolayer cell culture In vivo,changes in the microenviroment of the Space of Disse result in activation of stellate cells, deposition of fibronectin and production of cytokine activating agents by endothelial cells, and increased cytokine secretion and proliferation of Kupffer cells [52]. These results demonstrate that the presence of cell-matrix interactions is essential to the homeostasis of an in itroliver analog. 2) SolubleSinals Hepatic cells communicate via soluble signaling mechanisms following injury to induce the activation and termination of tissue regeneration [39]. Thus it is apparent that any in vitro system that aims for spontaneous regeneration or reconfiguration of sinusoidal structure must contain the necessary soluble signals. Though the exact mechanisms of these interactions has not been established, strong evidence exists that non-parenchymal cells play a critical role in the release and regulation of these soluble signals[53]. Because the composition and time dependence of cell signaling proteins during liver regeneration are unknown, supplementing media with the proteins seems more like a fishing expedition than a science, and a more Technology Massachusetts Institute Massachusetts Institute of ofTechnology 28 -28 - - Biotechnology Process Engineering Center Biotechnology Pocess Engineering Center natural approach is simply addition of physiological ratios of NPC's to in vitroco-cultures. However, for this to be done it is important that the various cell types from the liver by separable into their individual types, and fixed ratios (reminiscent of thoseseenin viv) of the various cell types then be added back into culture. A possible approach to do this is discussed elsewhere [54]. 3) Cell-Cell Interactions Cell-cell interactions, both homotypic and heterotypic have been shown to improve viability and function of in vitro hepatic cultures [43]. These cell-cell interactions consist of gap junctions, desmosomes, E-cadherins and tight junctions, who trigger a number of intercellular signaling mechanisms, which enhance homeostasis of cell phenotype [51]. Heterotypic interactions in hepatocyte-nonparenchymal cell co-cultures are thought to present a highly conserved signal that greatly augments liver specific functions [55]. Specific mechanisms that stabilize hepatocyte function have not yet been elucidated at the molecular level but homotypic and heterotypic interactions are thought to be imperative to the function and stabilization of the 'normal' liver phenotype [43, 45, 56]. 1.3 Metabolism of Xenobiotics in the Liver All organisms are exposed constantly and unavoidably to foreign chemicals, or xenobiotics, which include both man-made as well as natural chemicals such as drugs, industrial chemicals, pesticides, pollutants, toxins produced by molds, pyrolisis products in cooked food etc. The lipophilicity of the xenobiotics that enables them to be absorbed through the skin, lungs or gastrointestinal tract, is an obstacle to their elimination as lipophilic compounds can be easily re-absorbed. Consequently, the elimination of xenobiotics often depends on their conversion to water-soluble chemicals by a process known as biotransformation, which is catalyzed by enzymes present in the liver and other tissues. Biotransformation helps convert lipophilic compounds to a more hydrophilic form that enables their removal through feces or urine. Biotransformation helps speed up the clearance of toxic lipophilic substrates from the body. For e.g. the biotransformed form of hexobarabital Techno1o Massachusetts assachusetts Inswte Institute of ofTechnology 29 -29 - - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center is known to have a half life of 5-6 hours, while its lipophilic parent drug has a half life of nearly 2-5 months [57] Xenobiotics exert a variety of influences on biological systems, depending on theIR physicochemical property. This is of particular relevance to both pharmacology and toxicology. For e.g., in some cases, chemical modifications caused by the biotransformation of a drug may be responsible for the pharmacodynamic effect (or lack thereof) of the chemically modified drug, relative to the parent drug. In other cases, the modification may be responsible for the production of intermediates or final products that may or may not have a characteristic toxic effect or tumorigenesis effect that is possibly unseen in the parent drug. In most cases, however, biotransformation terminates the pharmacological effects of a drug and lessens the toxicity of xenobiotics. Enzymes catalyzing biotransformation reactions often determine the intensity and duration of action of drugs and play a key role in chemical toxicity and chemical tumorigenesis [57]. Xenobiotic biotransformation can thus be thought of as the principal mechanism for maintaining homeostasis during exposure of molecules to small foreign molecules, such as drugs. The terms biotransformation and metabolism are used almost synonymously, particularly in the context of drugs, though metabolism includes absorption, distribution, biotransformation and clearance - and is hence much broader in scope than biotransformation. The reactions characterized by biotransformation enzymes are generally divided into two groups, called Phase I and Phase II, as shown in Table 1-1 (From [571) Phase I reactions involve hydrolysis, reduction and oxidation. These reactions expose or introduce a functional group (-OH, -NH 2, -SH, or -COOH), and usually result only in a small increase in hydrophilicity. Phase II reactions involve acetylation, glucoronidation, sulfation, methylation, conjugation with glutathione and amino acids. The co-factor for these reactions with functional groups that are either present on the xenobiotic or are introduced/ exposed during Phase I biotransformation. Most Phase II reactions result in a large increase in hydrophilicity. Phase I and Phase reactions may occur independently or sequentially one after Massachusetts Institute Massachusetts Institute of of Technology Technology -30 - -30 - Biotechnology Process Engineering Center Biotechnolog Process Engineering Center the other. Xenobioiic biotransformation enzymes are widely distributed throughut the body, and are present in several subcellular compartments. In vertebrates, liver is the richest source of such enzymes. REACTION ENZYME LOCATION Phase I Hyvdrolysis Carboxylesterase Pep tidase Microsomnes,cvtosol, blood, lvsosomes Epoxide Hydroxylase M/Ncrosomes,cytososl Azo-and nitro-reduct ion Reducion (:arboml reduction Cvtosol Disulfide resuction Cvtosol Sulfoxide reduction Cvrosol Quinonc reduction Cvtosol: mcrosomes Reductive dehalogen:ation IMicrosomes AMcoholdehydrogena ase Aldehvde O.xidadtion l.icroflora, microsomes, cvtosol dehydrogei Cytosol nase Mkitochondia, cytosol Aldehyde oxidase Cytosol Xanthine oxidase Cvtosol Monoamine oxidase Cvtosol Diainne oxidase Cvtosol I-Hsynflthase lficrosomes Flavi-mniono-oxygcnases Ihlcrosomes Prostaglandin Cytochrome P450 Mficrosomes Phase II Glucoronide conjuga .tion Microsomes Sulfate conjugation Cvtoso Glutathione conjugal ion Cytosol, mnicrosomes Aninmo acid conjugat ion hfitichondria, microsomes Acvladon Mitochondria, cvtosol Methd4ation Cvtosol Table 1-1: General pathways of xenobiotic biotransformation and their major sub-cellular location 1Institute nstitUtc of Nlassachuserts Mlassachusetts of Technolov Technol(V, 31 -31 - Biotechnology Process Engineering Center Biotechnology Process ngincering Center Within the liver, they are primarily located in the endoplasmic reticululm (microsomes) or the soluble fractions of the cytoplasm (cytosol) of the hepatocytes, with lesser amounts in the mitichondria, nuclei and lsosomes. (Table 1-1) The presence of the biotransformation enzymes in the endoplasmic reticulum (ER) in the liver is possibly due to presence of the lipid bilayer in the ER. There is huge difference in the ability of different tissue to biotransfrom xenobiotics. This has huge toxicological implications, in terms of tissue-specific chemical injury. Several xenobiotics, including acetaminophen and carbon tetrachloride, are hepatotoxic due to their activation to reactive metabolites in the liver. [58]. Cells within the liver - depending on their location in the lobule also show considerable differences in their biotransformation of xenobiotics. For e.g. the cells in the centilobilar region are the ones that are responsible for the transformation of carbon tetrachlotride to their toxic intermediates. There are considerable differences in the distribution of the various biotransformation enzymes even with the same organ, across different species. [59]. The focus of this thesis will be on the biotransformation pathways mediated by the enzymes in the liver - specifically the Phase I CYP450's and some of the Phase II enzymes, as these have been known to be one of the most difficult to maintain in standard cultures in itro. 1.4 The CYP450 biotransformation enzymes Among the Phase I biotransformation enzymes, the CYP450 system ranks first in terms of catalytic versatility and the sheer number of xenobiotics it detoxifies or activates to reactive intermediates [60, 61]. The highest concentration of p4 50 enzymes is present in the endoplasmic reticulum in the liver, though they are also present in many other tissues. The p450's play a key role in regulating the intensity and duration of action of drugs, and they are also responsible for the detoxification of xenobiotics. They, play a key role in the biosynthesis or catabolism of steroid hormones, bile acids, fat soluble vitamins, fatty acids, and eicosanoids, and this underscores the catalytic versatility of the p45 0 's. Figure 1-6 details the key role of p450's in biology and medicine. technology Massachusetts Massachusetts Institute Institute of of'echnolSq ---------·- ·--- · ·- - 32 - - 32- ------- Biotcchnology Process Enneering Center Biotcchnology P-rocessEngneering Centcr All p450's are heme-containing proteins. The heme iron is usually in the Ferric (Fe 3) state. When reduced to the Ferrous state (Fe?2),the p450 can bind ligands such as oxygen and carbon monoxide. The resulting complex that forms absorbs light maximally at 450 nm. poiymorphisms drug desig antibiotics mutagen activation therapy design drug interactions promoters chemotherapeutics CARCINOGENESIS REACTIVE OXYGEN PHARMACOLOGY endocrine disruptors susceptibility growth actors V ENVIRONMENTAL pesticides TOXICOLOGY - ~ - P450 retrnoids - BIOTECHNOLOGY biomarkers OLOGY selective biocides \ ENDOCRINOLOGY phytosteroids adrenal seroids OLO HOMEOSTASIS androgens / | ecdysons\ estrogens REGULATION ENZYMES pollutants natural products neurosteroids juvenile hormone GENE neurostroids Yflower -- \ENDOCAIN \ \ \N faty vitaminD\ progesterones ketones color bile acids acids / eicosancids Figure 1-6: The role of p450's in medicine and biology The basic reaction catalyzed by the p450's is mono-oxgenation in which one atom of oxygen is incorporated into a substrate, designated RH, and the other is reduced to water, with reducing equivalents derived from NADPH as shown in Figure 1-7. Although CP450 functions as a mono-oxvgenase, the products are not limited to alcohols and phenols due to re-arrangement reactions [62]. During catalysis, CP450 binds directly to the substrate and molecular oxygen, but it does not directly interact with NADH or NADPH. The mechanism by with the p 4 50 receives electrons from the ND(P)H depends on the cellular location of the p450. In the endoplasmic reticulum in the liver, where most of the drug biotransformation p450's are located, the electrons are relaved from NADPH to the p4 5 0 via a flavoprotein (Figure 1-7) called NADPH-cvtochrome P450 reductase. flavoprotein electrons are transferred from the NADPH to the CP450 Initt fCehoov3 Mascuet Massachsetts Insitute of'Iechnolop,· -33 Within this via FMNNand FAD. itehooy-EgneigCnc Biotechnology Process Engineering Center In the mitochondria, that house the p450's involved in steroid hormone metabolism biosynthesis and vitamin D metabolism, electrons are transferred via twvoproteins - an iron: sulfur protein called ferrodoxin and and an FMN containing flavoprotein called ferrodoxin reductase. Phospholipids b5 also play an important and cytochrome role in the biotransformation function of CYP450 [57] Druq Oxdzed NHADP' H0O NADPH 02 Figure 1-7: Catalytic cycle of CYP450's The catalytic action of p450's (Figure 1-7) involves, in the first part, the activation of oxygen, and in the final part, substrate oxidation - i.e. the abstraction of a hydrogen atom or Maschsts Massacuserts Institue of Technology- nttueo Tcnooy-4-Bitcnooy - 34- ___ Cne rcssEgnern Biotechnology Process Engineering Center an electron from the substrate followed by oxygen rebound (also called radical recombination). Following the binding of the substrate to the p450 enzyme, the heme iron is reduced from the ferric to the ferrous form, by the addition of a single electron from the NADPH-cytochrome p 4 50 reductase. Oxygen binds to the p450 in its ferrous state and the FE 2 O, complex is converted to the Fe2-OOH complex by the addition of a proton (H) and a second electron from the reductase or the cvtochrome b5. This cleaves the ferrous complex to produce water and an (FeO)'3 complex, which transfers its oxygen atom to the substrate. Release of the oxidized substrate returns the CYP450 back to its initial state. CYP450 catalyzes many tpes of oxidation reactions including hvdroxylation of an aliphatic or aromatic carbon, epoxidation of a double bond, heteroatom (S-, N-, [-) oxygenation and N-hydroxylation, heteroatom (O-, S-, N-, Si-) Dealkylation, oxidative group transfer, cleavage of esters and dehvdrogenation. Detailed reviews on the chemistry of these reactions are available in literature [57, 58, 60, 62, 631. The relative abundance of the various CYP450's in the human liver (there are more than 400 different p450's that have been identified, and more than 59 pseudogenes), is shown in Figure 1-8. Figure 1-8: Relative abundance of the various p450's in human liver Studies on the biotransformation of new chemical entities by p450's leading to their activation/ inactivation, are one of a series of studies undertaken by the Pharmaceutical -gncigCne Mascuet inttt ofTcnlg M~ssachusetts nsitute of Tchnology 5-Boehoog - 35- rcs Biorechnolotgy Process ngeering Ce(:nter industry during the drug development process [1, 64, 651. In addition, information on which p450 metabolizes a drug can help predict or explain drug interactions [66]. For example, when administered with azole antifungals (e.g. ketaconazole) or macrolide antibiotics (e.g. ervtlhromycin), the antihistamine terfenadine (Seldane) can cause torsadesde pointes,which in some individuals has apparently led to lethal ventricular arrythmias [66]. This drug interaction can be rationalized on the basis that terfenadine is normally converted by intestinal and liver CYP3A4 in humans to a tertiary-butvl alcohol, which is further oxidized to a carboxylic acid metabolite. This latter metabolite does not cross the blood-brain barrier and terfenadine thus is a non-sedating antihistamine. When formation of the carboxylic acid is prevented by ketaconazole etc (CY'1P3A4inhibitors), the plasma levels of the parent drug terfenadine become sufficiently elevated to block cardiac potassium channels, leading to arrvthmias [66]. Thus competitive and non-competitive inhibition of CYP450's is an important screen for new chemical entities in the drug development process. In contrast to inhibitors, inducers of p 4 50 increase the rate of xenobiotic biotransformation [67-70]. Some of the p450's in the liver microsomes are inducible. Clinically important consequences of p45 0 induction include the enhanced biotransformation of cyclosporine, warfarin and contraceptive steroids by the inducers of CkT2C and CYP3A enzymes and enhanced activation of acetaminophen to its hepatocyte metabolite N-acetelvlbenzoquinoneimine, by the CYP2E1 and 3A enzyme inducers. Thus, in addition to CYP450 inhibition, CYP1450induction of new chemical entities is also screened for during the drug development process. In addition, mechanistic studies that determine possible drug-drug interactions, drug induced toxicity etc., involving a new chemical entity, is an essential part of the drug discovery process. 1.5 The Phase II biotransformation enzymes Phase II biotransformation reactions include glucoronidation, sulfation, acetylation, methylation, conjugation with glutathione (mercapturic acid synthesis), and conjugation with 36-36 Technology Massachusetts Institute Ma~ssachusetts nsitute of of Technology - - -I - · ___I - Biotechnology Process Engineering (:enter Biotechnology Process Engineering Center _·~~~~~ ... ... amino acids such as glycine, taurine and glutamic acid [71]. The co-factors for these reactions are shown in Figure 1-9. They react with functional groups that are either present on the xenobiotic or are introduced/ exposed during Phase I biotransformation. With the exception of methylation and acetylation, Phase II biotransformation reactions result in a large increase in the hydrophilicity of the parent compound. Glucornidation, sulfation, acetylation and methylation involve reactions with activated or high energy co-factors, whereas conjugation with amino acids or glutathione involves reactions with activated xenobiotics. Most Phase II enzymed are located in the cytosol, a notable exception is the UDP-glucuronosyltransferases, which are microsomal enzymes (Table 1-1). Phase II reactions usually proceed much faster than Phase I reactions, such as those catalyzed by CYP450 [57]. Therefore the rate of elimination of xenobiotics whose excretion depends on the rate of excretion depends on both Phase I as well as Phase II biotransformation reactions, is generally determined by the first one. Technology Massachusetts Mlassachusses Institute Institute of ofTechnology 37 -37 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center [ I.I ... z ... L.L~ 011 O IlO I ^-.u ------- I O·-I- _ I ounauo n (ouununj " 0-0-P-O-CH II 1 / o 3 ;Phosphoadenohne5'-phomphoulaW (PAPS) UrWdntw5'-dphoepbO-D-gIuJcronicdcid(UOP-GA) I~~r... Meoyllaton 1 r. OH H2N WOC",H I O CH-(CH,--S-C, H2 V 1OO ~~~~ Acerylcoem2yme A S-AdenoeylmethlonlneSAM) Amino Acid Conjugaion I Glutathlone Conjugation I I CH CG co SH, % V rglulImC aci . % cyMYlm * gIycwE r,lv3 I Glulthlos Taurine Glutmhee Figure 1-9: Co-factors involved in Phase II biotransformation reactions (fim [57]) Massachusetts of Technology Technology Institute of assachusetts Institute 38 -38 - - Biotechnology Process Engineering Center Biotechnology Process Engineeing Center 1.6 The Drug Development Process Discovering and bringing one new drug to the market tpically costs a pharmaceutical or biotechnology company nearly $800 million and takes an average of 10 to 15 years [1, 72]. Figure 1-10 is a schematic of the various steps involved in the drug discovery and development process. 4· ,$,; "... -IIllJII If u'.r Id, ,, Figure 1-10: A schematic of the drug discovery and development process New drugs begin in the laboratory with chemists, scientists, and pharmacologists who identify cellular and genetic factors that play a role in specific diseases. This leads to a search for chemical and biological substances that target these biological markers and are likely to have drug-like effects. It is estimated that out of every 5,()00 new compounds identified during the discovery process only five are considered safe for testing in human volunteers after preclinical evaluations [1, 72]. After 3 to 6 years of further clinical testing in patients, only one of these compounds is ultimately approved as a marketed drug for treatment. There is thus a high attrition rate of compounds as they step through each step of he drug discovery and development process, beginning with the demonstration of efficacy in experimental cell and animal models and concluding with the demonstration of safety and efficacy in humans. fTehooyMascuetsIsitt M~assachusettsInstirute of Tchnology· 39-BoehooyPoesEgneigCne - 39 - BiotcchiologWy rocess Frgine~ring (Center Drugs can fail at any point during the process - and failure at later times is inevitably more costly. Nearly 90% of the lead candidates identified by current in vitro screens fail to become drugs [5] ('igure 1-11). Many of the lead candidates fail due to unacceptable toxicity in one or more animal species. Among lead compounds that progress to Phase 1 clinical trials, a significant number fail due to unforeseen human liver toxicity [73] (Figure 1-11). This high rate of failure is inspite of an investment of more than 130 million dollars on predictive ADMET (absorption, distribution, metabolism, elimination and toxicity) screens, per $800 million dollars spent on developing a new drug. The projected annual five year growth rate in O-15°0o(rotre. PhRibLA) (Figure 1-12) expenditure on toxicology screens and assays is nearly 10% Why Lead Candidates Fail Market/Business Lack of Effica oxic ity 31% 22% Poor ADME 41% 90% of Lead candidates fail to become drugs Figure 1-11:The main causes of failure of lead candidates Nearlv 90% of the lead candidates entering Phase 1, fail due to liver toxicity [5] of Technology Institute of Massachusctt Institute Nfimsachusetts···" echno]M-g _·___ ___ 40 -40 - - Biotechnology Pnxess Engineering Center Biotechnology Process Engieering Ceter ____·__ In-vi $1.30 ADME Total Toxicology: $1.5 bn $1.50 bn In-vitro In-vitro $0.20 bn Surces: PhRNlABCC 2000/2001 $3.0 billion ir Growth Total 10% - 15% In-vitro >25% Figure 1-12: The growing expenditure on predictive in itro and in r'oanimalmodels of toxicology Thus, it is clear that there is an unmet need for a more predictive, cost effective screen of drug metabolism and toxicity in the liver. Indeed, there is considerable savings that can be had by failing chemical entities earlier in the drug discovery process - also called the fil early, /ail cheap paradigm.In addition, minimizing false negatives results in a significant opportunity' cost for drugs that may have otherwise not gone to market. By contributing to the understanding of a toxicity and providing perspective, investigational toxicology programs can enhance the discovery and deveklopment process. Investigational toxicology efforts can provide information regarding toxic mechanisms of a drug, and models for avoiding repeated toxicity problems. For toxicity revealed during preclinical development, determining the mechanism and providing an investigational model can contribute to the discovery or design of a less toxic analog. Alternatively, mechanistic studies can place the observed toxicity in perspective with regard to human risk. In mechanistic problem solvhing,it is important to select appropriate investigational models. While in i2wo models are needed to investigate drug effects in the context of toxicokinetics and systemic influences, in itr) models can be efficient and cost effective tools for investigating specific mechanisms in a precisely controlled environment. This is particularly so in the pre-clinical trial phase of the drug discovery and development process when there are far too many Technology MaacI1usert5 Mlasachusetts Institute nscture of of Technolog- -41 - - 41- Biorechnologv Process Engineering Center Biorechnolop- Procm En~ginecring Ceter compounds to be able to do animal testing, for pharmacology and toxicity assessment. The development of in itro model systems to evaluate the toxicity of chemicals and drugs has thus become increasingly important. These in itro models may also enhance our understanding of the mechanisms of drug and chemical induced toxicity, because in vivomodels are complicated by the presence of structural and functional heterogeneity and do not allow for mechanisms to be clearly defined or reproducibly examined. Additionally, in itro systems enjoy increasing popularity from the public because their application in toxicity testing may allow a decrease in the number of animals used in biological testing. In vitro models are usually target organ based and the most frequently encountered target organ toxicity is the liver. There are a number of in itro liver models such as perfused livers, primary isolated hepatocytes, liver slices, supersomes, microsomes, and transgenic cell lines - currently being used as predictive screens as well as mechanistic models of drug metabolism and toxicity (i.e as ADMET screens). However, its is pretty clear from the numbers in Figures 1-11 and 1-12, there has been little success in striking a balance between making the system easy to use and ensuring that the system recapitulates the most important functions of the in ri'o liver. Not all of the important functions can as yet be replicated at desired levels in in itro models, especially in culture formats amenable to routine use in drug development. In summary, there is need for an in litro liver model that is 'sufficiently' complex as to be able to replicate the liver pathology and biotransformation functions and thus fail toxic compounds early, while being amenable to use in a high throughput format. At the same time, the system must be precisely controllable and reproducible, to allow for its use in investigational toxicology, toxicokinetics and mechanistic xenobiotic biotransformation experiments. - technology Massachusetts Mlassachusetts Institute Instiute of of'lechnology - · _II_____ 42 - -42 - __ __·____·___ Biotechnology Process Engineering Cenrer iotechnology Process ngineering Cener 1.7 Hepatic Tissue Engineering for drug development assays: current status The type of in-vitromodel used to perform drug metabolism studies is a compromise between convenience and relevance [2, 4, 8, 65]. Figure 1-13, charts the various in vitroand in viv models used in the development of new drugs. Figure 1-13: In vitroand in vivomodels used in the development of new drugs, ranging from human to isolated enzymes, in order of in vim resemblance Taken from reference[3] Drug metabolism has been investigated previously in increasingly purified liver fractions culminating in single enzyme systems. In addition perfusion of the whole liver in-situ has been employed. The former reductionist approach suffers from lack of relevance to the invivosituation and the latter, while as close as possible to the liver in-vimo,suffers from complex methodology and limited multiple usage. Thus isolated hepatocytes and cultured hepatocytes may provide a convenient link between sub-cellular fractions and the complex architecture of the intact organ, while encouraging multiple experiments on the same batch of cells [8, 12, 65]. Thus in order to study liver function in a controlled environment, isolation and maintenance of viable hepatocytes is desirable. The use of an inherently simple system while maintaining the intact cellular architecture allows for accurate control of, for example, endobiotic/xenobiotic interactions, drug-dosing regimens, and the role of competing pathways in drug metabolism. Furthermore, a critical assessment of cellular function and dysfunction can be monitored in the presence of drug, inhibitor and co-factors. The models currently in use for human drug metabolism studies include [3] ns~ut ofTehnoogy-4 Masachsets Mvassachusetts Institute ofTechnologyJ - -43- ioecholoy rocssEngneein Cete Biotechnology Process Engneeing Center 1. Supersomes 2. Microsomes and cytosol 3. S9 fraction 4. transgenic cell lines 5. Primary hepatocytes and cultured primary hepatocytes 6. Liver slices 7. Perfused liver 8. In vivo animal model 9. Human Figure 1-14 summarizes the advantages and disadvantages of the various culture models. A detailed review of the advantages, disadvantages and future applications for each of the models listed above is available in literature [2, 3]. Technology Massachusetts Institute Institute of ofTechnology -44-44- Biotechnology Process Engineering Center Biotechnology Process Egieerig Center - In it,, tcimique H-nms CYP ad- U Advaupa I sit- Disadva upS_ - Osn isoyi Difflclt jurpaset Mxnpolam to HL2Mand m vivo Difflnmt goyple Highenzym activitis Humantivr macrtoms Unsuitable fr quantitative mersum Study of individual. gander-, and species- s Only CYP and UGTenzyes specfc bionnfomtiaa Human li cytosol NAT. Sr, and 0ST activ- Only NAT,ST; ad GSr ity depends on coafcts High Study zyuM activies gd'er-, of i'diduml and speci- spcific biotmnsfamtion Human li S9 faction Huma liv all a li Thnspnic ca lines Bol pas Low esqassima levels tIomnplre repmsntatia ef in vivo situation Only a lfw is eym expamu Study of om isaym or a af CYRP Well etablibhed and cha- Isdationcm be conplicatd andtm coming Study f mdiaton ad c- Only plSelocted cells can be studied zymc indatsn possible Ding tanspopmn still pmsan and opetionel hntac cellulr tractiom Moqtolgica studies pos- Liwrslics Cll duUs du gisdOimoD Indequate pnettima Damaed calls on de edges sibk Imendiv iduall viafim itmitd viable period canbe studied cExpesiveapquip ]solates perusd liver t Bie formatom Thro.didnsioal Limited viable peiod Po paduibility No human lier availabe Figure 1-14: Advantages and disadvantages of the various in itromodels Takenfirm refrences[2, 3] Technology Institute of Massachusetts Mabsachuserts Institute ofTechnology and cytosl Relatively stable enzym expression levels CYPs itducible Easy to culbs Hiihsr ssioD levels cambin Primy heptocylm Lwr amzyme activit than in misaoms I and I Study of individal gader-, and specksspecificbiotbmnsfction Easy to culum -45 -45 -- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Thus from Figures 1-13 and 1-14, its is apparent that primary hepatocytes may serve to be a useful model for a number of specific biotransformation studies, provided the hepatic function, specifically the biotransformation functions of the isolated hepatocytes or hepatocyte cultures are preserved in itro.Figure 1-15 summarizes the advantages and disadvantages of the use of primary hepatocytes or hepatocyte cultures are in vitr models. Advntages Wel established Well chmnclrized Virity fiir up to 4 weeks Study of madins andenzyme induces possible Disadvanulh Isolation ca be complicald andtimec... Vible cellenichmnt possible Cryopeaesvatii possible Drmu tnrorers still pesent andoprational Cellular nuctims Only pmselctd cels can be studied Call damage during isolation more difficult to study Figure 1-15:Advantages and disadvantages of the isolated hepatocytes and hepatocyte cultures as an in vitromodels Taken from references[2, 3] Although primary hepatocytes maintained under conventional culture conditions have been used broadly for short-term studies on drug metabolism and hepatotoxicity, the rapid loss of many liver specific functions, the failure to re-establish normal cell polarity and architecture, including bile canaliculi and the deterioration of cell viability within several days have limited their application for long term studies [74, 75]. Historically, three major complications have confounded the use of cultured hepatocytes for the study of drug transport and metabolism, as well as expression of liver specific genes. First, there is variable attachment and rapid functional deterioration of hepatocyte cultures maintained on plastic culture dishes. The second problem involves supplying the culture with adequate nutrients to carry out the large number and variety of cellular functions conducted by hepatocytes in-situ (e.g. synthesis of serum proteins). The third problem stems from an incomplete understanding of the hormonal requirements of the cultured hepatocytes. Whether due to one or a number Technology Massachusetts Institute Massachusetts Institute of ofTechnology -46 -46 -- Biotechnology Process Engineering Center Biotechnology Process Engineering Center of these factors, primary cultures of rat hepatocytes inevitably show a complete loss of greatly diminished activity in liver specific protein synthesis [76, 77], xenobiotic-metabolizing activity [78], cytochrome p450 induction [79] and Na+ linked bile acid transport [8] within the first 2448 hours when maintained under conventional culture conditions. Further, the failure of the cultured hepatocytes to re-establish normal bile canalicular networks (cell polarity) has also limited their application for examining the regulation of biliary secretion and mechanisms of choleostasis [8]. These restrictions have been a major obstacle for performing studies pertaining to the metabolism of drugs in as much as cytochrome p450 mediated metabolism frequently determines the toxicity and mutagenicity of many natural and synthetic chemicals. In addition the loss of cytochrome p450 and other drug metabolism enzymes limits the kinds of studies that can be performed with the primary hepatocyte cell culture models, and raises uncertainties about the relevance of findings in cultured cells to the intact liver. Initial attempts at culturing rat hepatocytes were done using simple or conventional culture conditions: seeding individual cells in static medium in dishes coated with extracellular matrix proteins such as collagen I or matrigel. Some methods involved re-suspending cells in physiological medium containing serum and growth factors and plating them onto rigid substratum such as plastic or protein coated dishes. Under these conditions, hepatocytes spread extensively, assume a flattened morphology and eventually form a confluent monolayer. Rubin et al [8] demonstrated that hepatocytes attach more readily to a substratum of gelled collagen I than to a rigid substratum of the same material. Many other studies have been performed on different culture systems including the immensely researched 2D sandwich configuration wherein hepatocytes are cultured in-between two successive layers of ECM [18, 24, 50, 80]. Lecluyse & co-workers have extensively researched the possibility of growing hepatocytes in matrigel [80] while Wu and co-workers have used suspension cultures to form spheroids [20]. These cultures have been shown to maintain hepatic function (serum protein secretion and p450 induction) for extended culture periods. However, spheroidal cultures differ structurally from what is seen in-vio. The lack of perfused flow through the spheroidal aggregates prevents nutrient transport to the cells in the interior of the aggregate, especially in case of spheroids whose sizes exceed diffusion length scales. A detailed review of the various Technology ofTechnoloy Insliture of vIassachusetts assachusetts Institute -47 -47- Biotechnology Process Engineering Center Biotechnology Process Engineering Center in vitro primary cultured hepatocyte models and their drawback are discussed in detail in literature [4, 8, 65]. In general, none of the current in vitrocell culture models have been able to replicate all the pharmaceutically relevant functions of the liver [80]. In any culture system it is necessary that an environment be provided which ensures long-term maintenance of hepatic function and cell viability. Adequate nutrient and oxygen transport is critical in the maintenance of cell viability over extended days in culture. At the same time, it is important that many of the facets of the in ivomicroenvironmental cues - cellcell homotypic and heterotypic interactions, soluble and shear mediated signalling, and adhesion guided cell-matrix signalling, be provided to the primary hepatocytes in culture to replicate in ivo like function. At the same time, it needs to be borne in mind that all of the in vitromodels discussed in Figure 1-14, may have very specific uses for a defined set of biotransformation experiments. An excellent review by Brandon et al.[3], summarizes possible uses for some of the standard in ito models. This is reproduced in Figure 1-16. Indeed, as suggested in the figure, stable, iin im like cultured primary hepatocyte models may be poised to be a very useful screen for testing the toxicity of a drug and its metabolites. Technology Institute of Massachusetts MassachusettsInstitute ofTechnology -48-48- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Drugrsecb Inviu modl Axgumcuts Drmgbiransoumiaion (1) Pooled microsoms ad (1) Combination ezyrns in one model. CYPs and UGTs in micrusana and soluble phase 1 enzymes in cytosol (enr-speific fiaicuos can be used) (2) Supmomes and NAT (4) (rmgargic) cell li -rimary hepatocyes (5) lver sties Isolation mtabolis ptsme (3) Combiation of phase I and I ezymes (4) intact ci to study (3) Human liver S9 frcia and (2) One spcifi isozy (6) Ntfmed anim liver (1) CYP or UGT supera NA cytosol or sa (2) Milrosomes or cytoal (5) Intact ivhNrstu (6) Only for bile cmretion (1) Mctabolim of a specific isoyme ld) (hiher (2) Only whe isoeazynt is not psent as isodaed en- zym Drug-drug intracticm IIbi.cntfntia (3) Only wbho m (3) S9 fIacti (1) Microsomes or cytsol C-) CYP or UG super- aetbdiis canbintim (1) Combinaftionof (2) of pse I and zynms in ono modl at aom specific isoyme diton sam ar NAT cytod Influen of polyme- (3) PRimay hbetocyles (3) ntact cell (1) upsnoms (1) CYP p (2) Micnuoines or cyteac (2) Prom one paent that shows that polymor- (3) Pimary hqepatocys (1) Pimayhepaocyes (3) Fn om patient that shows that polynmphism (1) htact cell and cytetacicity is nmasurable C) Clhmannic) Ci (2) Influe phims odpbism in supenomes available phism Toxicity of drug and its mtabbdhtes cytotaxi of on or combinaion f enzymes can be studied (3) inact liw suctme (1) Dimr model to1ol (3) Superslms (2) Rat CYP supears (3) S9 fatio (4) Phnuly t animal modls ae aailable mpxrocytes a- available (3) Diffr nimalmods an available (4) Iutactcell and differnmt aimal modelhs- available Figure 1-16: Applicability of current in itromodels in various stages of biotransformation research 1.8 Hypotheses and Specific Thesis Objectives This thesis will be centered on the hypothesis that the higher order functions of the liver, including its biotransformational capacity can be best recreated and preserved in an in vitroculture, if the critical facets of the in ivo microenvironment namely - cell-cell signalling through homotypic and heterotypic interactions, adhesion guided cell-matrix signalling and Massachusetts Massachusetts Institute Institute of of Technology Technology -49 - -49 - Biotechnology Process Engineering Center Biotechnoloy Process Engineering Center soluble and shear mediated signalling in a three dimensional environment - are recapitulated in the in vitroanalog. The specific objectives of this thesis are: 1. To determine appropriate design parameters to create a three dimensional "in vim" like "in vitro" system - specifically, to maintain crossflow through the tissue and to optimize the crossflow rate to replicate in im shear rates, while satisfying tissue oxygen demand. 2. To assess the ability of the system to maintain liver function - with emphasis on the biotransformational capacity of the system - using broad metrics of gene and protein expression and biochemical activity. 3. To use this in vitro system to extract quantitative rates of metabolism of a candidate xenobiotic. 4. To compare the biotransformational capacity of the designed system with that achieved by standard cell culture models used in the pharmaceutical industry, again using metrics of gene and protein expression and biochemical activity. 5. To model the disposition of any chemical species (including a xenobiotic) in the system taking into account appropriate mass transfer considerations. Massachusetts Institute of Technology -50- Biotechnology Process Engineering Center Chapter 2 Design and Fabrication of the 3D Microfabricated Liver Bioreactor and Fluidics As described in detail in Chapter , there is an unmet need in the pharmaceutical industry for a predictive in tro hepatic tissue culture screen that may be used in pre-clinical hepatic drug metabolism and toxicity studies. As described for the specific case of liver tissue models in Section 1.3, in itro models of heterotypic cell interactions are essential tools in dissecting dynamic physiological and pathophysiological processes. Such models fulfill an important connection between well-defined cell cultures and the complexity of the whole animal. They also provide experimental models of human tissue responses, where in 'il'o models are usually unavailable. Most primary liver cell culture systems currently available have shown rapid loss of liver-specific function and failure of the cultured hepatoqctes to reestablish normal bile canaliculi, cell polaritr, and cell architecture [4, 8, 11, 65]. One specific goal of this thesis was to develop a facile method for recreating a perfused capillan, bed structure of liver tissue in titr as a tool for studying liver physiology of particular relevance to the Pharmaceutical industry. 2.1 Key facets of the in vivo microenvironment that need to be recapitulated in an in vitro system Most tissues comprise a hierarchical arrangement of cells penneated by capillary blood vessels. Tissue homeostasis is maintained in part by communication between the different cell types in a tissue. Each cell receives signals from its neighboring cells via direct cell-cell interactions, cell-matrix interactions, and via soluble signaling molecules (cytokines and growth factors). In addition, mechanical forces -- such as shear stress on the endothelium from flowing blood -- are converted to chemical signals that are necessary for normal tissue function [81-841. Techno1ov Insdture Massachusetts NfassachustettS Institureof offlechnolop, 51 -51 - Biotechnology Process Enneering Center iotechnolgy Prorcess Rngineering Center The in ivo microenvironment (Figure 2-10 of hepatocytes includes signaling mechanisms mediated by cell-cell and cell-matrix interactions, soluble factors, and mechanical forces. Primary liver cell isolates contain hepatocytes and non-parenchymal cells (stellate cells, Kupffer cells, endothelial cells) and can be induced to recreate features of liver tissue structure in itro [17, 22, 27, 55, 85, 86]. What is a 3') Perfused Liver Analoq - Minmicin vivo 30 Tissue CON. vein artery 1 ,ll macrophag6e .vlI'. (Kupffer cell) * enable physiological 3D cell-cell relationships of NPC and hepatocytes Figure 2-1(a): Important facets of the in tivo microenvironment that need to be recapitulated in an in iitm system Need to capture the histotypical interactions between the various liver cell types. Although a rich array of two- and three-dimensional co-culture models have been developed, the available models lack one crucial feature of most tissues -- a perfused microvasculature - Figure 2-1 (b). Unlike larger vessels, which can be attached to a flow system, Instute of Massachusetts Istiute Mlassachusetts of Technology Technolog- a- - - 52 -52 - Biotechnology Process Tngineering Center Biotechnolog Process Engi~eering Center the small size of capillary blood vessels make them inherently difficult to perfuse in itro in the context of host tissue.. As a step towards creating physiological mimics of human and animal tissues that recapitulate the features of a capillar bed, an in itro sstem that facilitates perfusion of 3D heterotypic co-cultures at the length scale of the capillary bed in an arrangement that also allows in situ analysis of cell behavior via microscopy and spectroscopy, is needed. What is a 3) e Liver Analog - Mimic in vivo fluid/cs .-;' .IV d; as ;,le !%,ud mi I- c-'harmc i . - I The Lobule- a hexagonal repeat unit A 4; '- . 4:L I,A lY!'E9lJ 511, f" 'X Sx - tissue v 4 l"N- '11 _' '!~! i~V o'3~,,, ~rwr~ ,l~ Iki r PORj4 av e l 's"m''. I, , L . Se vein ,IuVjL_ Pnlsrsjsog) C PE artery . enable controlled perfusion through tissue: interplay of biology and fluid mechanics shear-induced signaling by endothelium I U woidg central vein Figure 2-1(b): Important facets of the in it'o microenvironment that need to be recapitulated in an in ritro system Enable controlled perfusion through the hepatic tissue 2.2 Design principles:biophysics of tissue morphogenesis A mixture of isolated cells of many different types has an intrinsic propensity to reorganize in a histotypic fashion (where cells of the same type come together) into a finctional tissue, if provided an appropriate length scale and time scale to re-organize [55, 85]. nl g ~ M saettsss a~c htu~t o.Tc 'Alassachusetts Insitute of Technoloy .nt . - 53- i tcnl g r cs n n e i g C ne Biotechnolyy Process Eninering (enter This was well illustrated in a series of landmark experiments by Steinberg and co-workers [85] and more recently by Michalopoulous and co-workers [21, 22]. In addition, the differential adhesion of cells to matrix substrata, has been shown to be an important consideration in the morphogenesis of different cell types into a tissue [55, 85, 87]. The biophysics of cell-cell cohesion mediated through cadherins (calcium-dependent transmembrane proteins that interact homotypically with their counterparts on neighboring cells), and that of cell-substrate adhesion through integrins ( heterodimeric transmembrane proteins that recognize and bind mainly to components of the extracellular matrix) have been shown to be important determinants of ultimate morphology, cytoarchitecture, and organization achieved by a mixture of different cell types in itm [55, 85, 87, 88]. Principles of In VitroMorphogenesis cell-cell ,'~~0 P~i "~adhesion ;m receptor mix relativestrengths of adhesion t Cell-Cell Forces > Cell-Surface Forces igh_ _ _ a~w uffiihx nffiritu day Intermediate (examples) high b _ la ; -ffinitf affinity Cell-Surface Forces > Cell-Cell Forces high_ _ ola bigP_ _ _I; ffiriv ISteinberg, a-liritv : ity. .ff.n.t.................... ..!fjn. ,,; M. (1963) Figure 2-2: Cell-Cell homotypic interactions that lead to histotypic re-organization over a defined length and time scale and adhesion guided morphogenesis of pure and mixed cell populations. The differential substrate adhesion strength of the cells A and B is shown to be an important determinant of tissue morphology. 54 -54 of Tcchnologv Institute of Massachusetts Institute Massachusetts T'cchnolop, - ""-- - Biotechnology Process Enneering Center .Biotechnolog Process En~ineering Center "`- Thus the design of an appropriate in itro system will involve the creation a 3D environment that will facilitate tissue morphogenesis in a controlled fashion - i.e. by engineering a system that affords a control over the homotypic associations between similar cell types, over the heterotypic associations of different cell-types and over the adhesion guided biophysical interactions between the cells and their matrix substratum. The key principles in tissue morphogenesis are summarized in Figure 2-2. The scope of this thesis was limited to engendering the homotypic interactions between the hepatocytes and the cell-matrix interactions between hepatocytes and collagen I in a 3D environment and studying the biotransformational capacity of the tissue so formed. Two important components of the in ivo microenvironment - the replication of the heterotypic interactions between the hepatocytes and the other cell types in the sinusoid (the endothelial cells, stellate cells and Isuppfer cells) by including physiological ratios of the various cell types, and the control over soluble signalling mechanisms - were not addressed as part of this thesis. While the non-parenchymal cells are possibly present in the cultures prepared as part of this thesis, they are possibly present at subphysiological ratios with respect to the parenchymal cells. However, the bioreactor design is sufficiently general in being able to incorporate these aspects of the microenvironment in future experiments. 2.3 Microscopic design parameters Key issues that affect the microscopic design parameters of the in vitrosystem include constraining the size of cell aggregates (controlling the length scale to allow for histotypical reorganization and to prevent nutrient depletion), controlling the uniformity of aggregates (to ensure repeatable cultures), and obtaining a physiological perfusion rate through 3D tissue structures (to simulate physiologically relevant mechanical stresses in the system). As a first step in this direction, a microfabricated bioreactor that comprised an array of 40-100 capillary bed-sized (-0.23mm x 0.3 mm x 0.3 mm) channels in a silicon chip and a reactor housing that enables a stable cross-flow perfusion of cells and cell aggregates seeded into the channels, was developed. The reactor was designed to enable long-term 3D culture and to allow in situ optical imaging (via 2-photon microscopy) and spectroscopy of the three dimensional cultures to assess structure and function. The heart of the reactor is the scaffold, comprising a thin (t - Inttt ofTehooy-5 Mascsts Massachustts Institute of Technology -55 - itcnlgyPoesEgneigCne Biotechnology Process Egneering Center 230 mrn) silicon sheet permeated from top to bottom by a regular array of channels and seated above a micro porous filter, which in turn is mechanically supported by a second silicon scaffold. The channels in the upper scaffold hold the liver cells and influence morphogenesis via the nature of cell adhesion to the channel walls that is coated with extra cellular matrix (thus influencing cell-substrate adhesion), and via the physical dimensions and geometry of the channel cross section (thus influencing cell-cell homotypic and heterotypic interactions). Perfusion rates are to be optimized taking into account both oxygen mass transfer and shear stress considerations. A square cross section of 0.30 mm was chosen to allow for a sufficient length scale for tissue re-organization [55, 85, 87, 88]. Figure 2-3 details the microscopic design parameters of the bioreactor. The width of the channels in the bioreactor was set to mimic the appropriate length scale needed for histotypic cell sorting. The channel depth mimics the length of the hepatic acinus. The low hydraulic permeability of the filter allows for a large pressure drop across the filter enabling a uniform crossflow through every channel of the bioreactor independent of the tissue distribution and cell numbers in each channel of the bioreactor. The axial flow rate was set to a high value of 0.5 ml/min to minimize oxygen concentration gradients along the length of the bioreactor (Pe - 100 >>1). This thesis focused on the maintenance and optimization of the perfusion or crossflow through the tissue in the channel of the bioreactor. The crossflow rate (and hence the values of the Peclet and Reynolds numbers for the perfusion flow rate through the tissue) was optimized taking into account the twin considerations of simulating physiologically relevant shear stresses while also satisfying the tissue oxygen demand. Massachusetts Institute of Technology -56 - Biotechnology Process Engineering Center . p... F L L: Microscopic Reactor Design Parameters channel dimensions and crossflow rates . ' .b. . I I .1 ~- - .. ~~" - - - I 11I. Filter 5 pm pore: ; (low hyd.Permeability) I s - QperfliO channel w: . . s . . . ' ; w 1 9-224 fit 10 ... APfilter hit f-hannels length scale of cell sorting and self-organization = 300 urnm (Powers, 1997: Steinberg, 1963) t - 30 um (mimic length scale of lobular repeat unit) Qxflow ~ to be optimized based on simultaneous considerations of tissue oxygen demand and the need to simulate physiologically relevant shear stress Qin - 0.5 ml/min, so that Pe >> 1, negligible axial oxygen concentration drop along the length of the reactor Figure 2-3: The design parameters used in the fabrication of the cell-holding channels in the silicon scaffold. The tissue perfusion and bioreactor axial flow rates are set taking into account the need to simulate physiological shear stresses in itro while satisfying the tissue oxygen demand 2.4 Photolithography: fabrication of the silicon scaffolds All scaffolds used in this thesis were fabricated in house bv Dr.lKarel Domansky at the MilcrosvstemsTechnology Laboratory at MIT. Scaffolds were fabricated out of silicon. Silicon allows facile prototping of a variety of channel cross-sectional geometries and dimensions. The silicon scaffolds were fabricated by deep reactive ion etching (DRIE) of double-side polished 230 m thick <100> boron-doped silicon wafers. Because the DRIE processes exposes the patterned wafers to SF6 etch followed by passivation in C4F8, the sidewalls of the microchannels are covered with a thin laver of a fluoropolymer. In contrast, the top surface of the silicon wafer is devoid of the fluoropolhmer due to the protection by photoresist during the DRIE process (igure 2-4). .s Inttt fTcnlg [aschst Masssachueits Instiute of'lechnolop- itcno yPoesEgneigCne Biotechnolop Pocess Eninering Cener MrOOK01111DITKO lo nhotoresis '. E.,!J ! . 'Jllj . I I18 }It i It i '.}.8 t! ; 11i ;:.-, iAsil'e u f iip dl It-'c.b i . I' l )CI . "n jqt{eC: rqqffi - t it RI . I~ e Ad ''1 A singlechanneledge ; ':Si ., . il q, 1 Channel Geometries pnotoresil_ !4-:.lt ! l-II- . .I· . .I.!;:.1.! ! quartz . : I!: .;I - I - simple - 1".- I complex Figure 2-4: Schematic of the DRIE process (left) used to fabricate the scaffolds and photos of the channel structure and geometries showing resolution. Upper photos show a light micrograph as well as an SEM (Scanning Electron Micrograph) of the scaffold structure (adapted from figure in [891) Because the system is designed for the formation of microtissue units within the channels, it is essential that cells can attach to the channel walls while remaining non-adherent to the top oxidized silicon surface. Previous results from experiments conducted in the lab, suggest that the cells adhere more strongly to the collagen coated photopolymer surface than to the collagen coated silicon dioxide surface [89]1.Silicon scaffolds with channels and slits of varied geometry are showed in Figure 2-5. Technology of Technollop, nstitute of 'Massachusetts assachustts Institute - - 58 -58 - - Biotechnology Process Engineering Center Biotetchnology Process Frigincering Center Figure 2-5: Silicon scaffolds of different geometries microfabricated by DRIE (Photqgraphby Dr.Karel Domanwskj) 2.5 Design and fabrication of the polycarbonate bioreactor housing The bioreactor housing features top and bottom polycarbonate compartments, each with flow inlets and outlets. The scaffold assemblv fits into the 12 x 7 x 2.4-mm oblong pocket in the top compartment (Figure 2-7). The bottom compartment functions as a lid enclosing the bioreactor. A Buna O-ring with a 316-stainless steel retaining ring provides the seal. Both compartments are approximately fabricated b micromechanical milling and thermal diffusion bonding. The bonding technology, 24.5 x 16 x 3.1 mm in size. The top compartment originally developed for PILMA [90), 91], was modified b was Dr.Karel Domanskv and co-workers for polycarbonate [92], tested on a batch of prototqypes, and transferred to Eastern Plastics Inc. (Bristol, CT) for fabrication. The reason for using a bonding technique instead of an adhesive was to avoid the possibility of leaching chemicals harmful to the cells into the cell culture medium. Prior to bonding, features in the compartment were micromachined on both sides. The 800 x 400-xrn surface channels were closed (forming Dshaped channels) by the thermal diffusion bonding of a thick sheet of polycarbonate to the rnicromachined part and lapping it to 125 ,urn. The dimensions as well as the curvature of the [)-shaped channels were set based on experimental results obtained from tissue cultures rumn on a version of the reactor called MilliII. The poor tissue phenotqpe in the MilliII reactors resulted in modifications to the design of the inlet chamber to minimize the possibility of cell stress/ shear when multiple spheroids of 3001Lmdiameter entered the inlet port simultaneously (Figure 2-6). In this way, a thin, optically clear window for microscopic observation of the cells Institute of Massachusettss Institute Nfasiachusettt of Technology Tetchnology~ - 59 - 59 - - Biotechnology Process Engineering Center Biorechnolop, Process Engineering Cener was formed. The size of the oblong fluidic chamber above the cell-holding scaffold was 8 x 3 x 0.725 mmn.The bottom bioreactor compartment was fabricated by micromechanical milling. The threaded fluidic connectors were custom-machined from polvetheretherketone (PEEK) and sealed with Viton O-rings. The internal volume of the reactor (including connectors) is 102 l. Lessons learned from Milli HIexperiments - Valuable inputsfor design of next prototype Necessary Changes to Design: nf c-hin fnr length occRnvx withnut . MP-rhnnirmn tn icnlatp ollc in entrance for a fullyIncdeveloped flow - lMarginalincrease · Marginal increase in entrance length for a fully developed flow t · Increase in inlet port size - from 300 .tm to 800 gm · Chip geometry - round vs. oval - minimize misalignment, "hands free" assembly of reactor * A gradual change in flow direction into and out of the reactor chamber - minimize cell shear Figure 2-6: Changes made to the design of the MilliF reactor, based on feedback from experiments on Mill reactors Technology Institute of MassachusetrsInstitute ofecnology~ 1M~assachusetts __ 60 -60 - Biotechnology Process Engineering Center iotechnology Process Engineering Center - __· Silicone gasket Silicon scaffold :affold Microporous filter ssembly ¢,... Silicon scaffold . . .-. '~.. Silicone gasket Q-----/ 0-ring with 9 , bonded by thermal diffusion retaininginsert Figure 2-7: An expanded view of the scaffold assembly left) and a schematic of the various parts of the bioreactor (Schematicand photograpbs1?,Dr.Kirel Domansk) 2.6 Assembly of the bioreactor Polvcarbonate reactor compartments and polycarbonate scaffolds were sterilized in 70% ethanol. All other reactor parts, tubing, connectors, and reservoir were sterilized by autoclaving. Cell scaffolds were coated in 30 g/mL Vitrogen (type I collagen) (Cohesion Technologies, Palo Alto, CA) for 30 min and rinsed in phosphate-buffered saline (PBS), pH 7.4 (Life Technologies, Rockville, MD). Hydrophilic Durapore M T filters (fillipore Corp., Bedford, NL) with a pore-size of five microns were punched with a custom-made oblong in PBS. puncher and coated with 1o bovine serum albumin (BSA) (Sigma, St. Louis, MO10) Components of the scaffold assembly were individually inserted into the bioreactor pocket as shown in Figure 2-7 and secured by attaching the bottom reactor compartment using three screws. Four threaded holes in the corners of the reactor were used for screwing Tnrtt ofTcnlg \issch-et MLassachustts nsitute of Technology 2-BoehooyPoesEgneigCne Biotechnology Process Enineering -61- C:enter the reactor to a plastic mounting bracket. A photograph of an assembled reactor is shown in Figure 2-8. .!i~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~ s N II. ~ .v. 11W~~~~~~ .4 .:~ Figure 2-8: Photograph of an assembled bioreactor (Photographby 3 Dr.Karel Domansky) 2.7 Design of fluidic system A single pump fluidic system was originally designed to go with the bioreactor. The bioreactor was connected to a 40-ml polyvpropylene reservoir with silicone (Silastic(®,Dow Coming, NJ) tubing. The reservoir featured a customized lid with fluidic feedthrough connectors. A mini peristaltic pumps (Instech Laboratories, Inc, Plymouth Meeting, PA) equipped with C-flex® pump tubing of different internal diameters for selected flow rate ranges were used to circulate the culture medium as shown in the schematic in Figure 2-9. The crossflow rate was determined exclusively by the pressure drop across the filter, sandwiched between the two silicon scaffolds. Thus, a portion of the axial flow was pulled through the channels of the silicon scaffold in the bioreactor and eventually went into the reservoir. The system was operated in complete recycle. '1'cchnology Intiwre of N1assachusett Tnititue Maslsachsetts of'lechnology 62 -(62 - - _____ Center Biotechnology Process Engineering Center Biotechnology Process Ennecring Single Pump Fluidic System -.I ~. ---I imaging & spectroscopy . # reservoir Figure 2-9: Single pump fluidic system used to run culture media through the bioreactor 2.8 Maintenance of crossflow in the bioreactor During the initial runs of the bioreactor, the perfusion flow through the silicon chip was observed to dnr up within 24-48 hrs. after seeding the reactors (Figure 2-10). It was hypothesized that this occurred either due to: A: Increased impedance in the cross flow line due to bubbles in some channels of the bioreactor. B: Clogging of the Durapore membrane filter in the scaffold sandwich by cell debris and spheroids. Masachuserts MassachusetsIntirntc Intitte of of 'Iechno]ogx' T~'chnolqgy - 633 - - - I3iotechnologv Process Engineering Center Biotechnolopr Process Engineering Cnter Crossflow measurement in Hepatocyte Culture 0.08 0.06] 3 o 0.04 ', ,. I 0.02 - Ca 0 -n 70 10 20 30 40 50 60 Cells seeded Time (hrs) post seeding Figure 2-10: Loss in crossflow seen in 3D bioreactors 24-48 hrs. post seeding In order to test hypothesis A, a second peristaltic pump was used in the cross flow line to maintain a constant as shown Figure 2-11 below. It was reasoned that the introduction of the second peristaltic pump (that maintains a constant flow independent of head or impedance, so long as the impedance is below the rated head of the pump) in the crossflow line would help maintain a constant flow in the line. Axial Flow 500 ld/min Reservoir To I Reactor From Reservoir ................... Reversed flow 75 pl/m in Figure 2-11: Schematic showing a two-pump fluidic system where the second crossflow pump pushes fluid from the top of the reactor in through the channels 6 -64- MasahuetsnsittcofTehnloy Massachusetts Institute of T'echnolog - -'--- I BorcholgyPrces ngnerig ene Biotechnology Process Engineering Center However, the cross flow was found to dry up even in this case. This suggested that hypothesis B, maybe the most likely reason for the clogging of the filter. Another related set of experiments conducted simultaneously, wherein the flow in the cross flow line was reversed, showed that the reactors started leaking in the crossflow line within 24-48 hrs of reversal of flow, possibly due to the clogging of the filter in the reverse direction, in the absence of any filter in the external flow circuit to collect and screen the cell debris. The next set of experiments involved the use of two filters (marked F) in the external flow circuit as shown in Figure 2-12 below: Axial Flow To Reservoir 500 gl/min Reactor F1 - From Reservoir .. ,*······· v,_,/ 75 gdl/min (A) Reversed flow ...................(B) Normal flow Figure 2-12: Schematic of the two-pump fluidic circuit after the addition of the inline filters Filter F (0.2 p.m) is added to the axial line of the reactor during seeding and is removed within two hours after seeding. The purpose of this filter is to remove most of the excess spheroids that did not go into the channels. This helps eliminate any noise in the albumin data due to secretion from suspended spheroids / spheroids in the flowing medium. Immediatelv after seeding, the pump in the cross flow line can either be connected in normal flow mode (in the direction of arrow B, as shown). After 24 hrs, when the spheroids are well spread and have formed structures, a filter is added to the crossflow line (F2, 0.8/0.2 pm Supor* double filter from Pall Gelman), and the direction of cross flow is reversed. In reversing the direction of crossflow, adequate precautions need to be taken to: 1. The crossflow line as well as the bottom of the reactor must be primed to minimize air bubbles that can. jostle the cells out of the channels. The fourth port (not used in normal reactor operation) may be used to prime the CF line using the crossflow pump i.e. keep the fourth port open till the crossflow line is primed. Use of smaller diameter tubing in the Masacustt Manssachusetts Insdrutc of Tchnolop, I~uut 6 -Bitehnloy o Tchnloy -65 - ioechnolopr PocssEninerngCete PI-rocessE~ngireerig enter crossflow line helps minimize the time needed for priming the line. Moreover, use of a syringe to prime the line helps minimize the time needed for priming. 2. The filter used in the crossflow line must be passivated with the culture medium to minimize nutrient depletion in the flow circuit. . , . Figure 2-13: Cell debris seen in the media in the reservoir 1.5 hrs. after reversal of crossflow Figure 2-13 above is a picture of the cell debris and unattached spheroids collected in the medium (taken 1.5 hrs after flow reversal). The presence of filter F2 prevents this debris from entering the crossflow line and clogging the DuraporeR' filter placed between the silicon scaffolds. It is a good practice to change the medium in the crossflow line, 2-3 hrs. after flow reversal. Moreover, if possible the filter F2 in the CF line should be changed whenever the media is changed - once evetr three days to prevent clogging of the inline filter leading to a loss in crossflow. The crossflow is maintained at a constant flow rate for the entire duration of the culture, by using the scheme described above. Figure 2-14 below shows cell debris being removed from the filter as a result of the reversal of crossflow. The tissue structures however remain intact in the channels of the bioreactor. - - -~~~~~~_ roessEngncrin Cete 6 - 6- nsttut o Tehnoog Masachsets Nla,,;sachsetts Institute of'l'cchnol(V- - __ ~~ ~ ~ _ _ -Bioecnolgy Biotechnology P'rocess Enaincring Center ~ ~~~~~~~~~~~~~~~~~~ _ - -__ (a) (h) Figure 2-14: Cleaning effect of crossflow (a) Before reversal of crossflow (b) 15 minutes after reversal of crossflow Thus, a modified design of the fluidic circuit optimized for use with the bioreactor, that ensured the maintenance of a constant perfusion rate (crossflow) through the tissue, is shown in Figure 2-15. imaging & spectroscopy vent filt Figure 2-15: Schematic of the optimized two-pump fluidics used with the bioreactor Technology Massachusetts Mlasschusetts institute Insrituteof ofTechnology - 67 6 - - Biotechnology Process Engineering Center Biotechnok)~T, Process Eninering Center 2.9 Summary of key results and conclusions A microfabricated bioreactor system that allows for three dimensional morphogenesis of liver tissue under continuous perfusion conditions was developed keeping in mind important facets in titr microenvironment that had to be recreated in the in itro system. A key feature of the bioreactor is the distribution of cells into many tiny, (-0.001 cm3) tissue units (capillary bed-sized (-0.23mm in a silicon chip) that are x 0.3 mm x 0.3 mm) channels uniformly perfused with culture medium. The total mass of tissue in the system is readily adjusted for applications requiring only a few thousand cells to those requiring over a million cells by keeping the microenvironment the same and scaling the total number of tissue units. The reactor was designed to enable long-term 3D culture and to allow in sitt optical imaging (via 2-photon microscopy) and spectroscopy of the three dimensional cultures to assess structure and function. The channels in the upper scaffold hold the liver cells and influence morphogenesis via the nature of cell adhesion to the channel walls and via the physical dimensions and geometry of the channel cross section. Perfusion rates are optimized taking into account both oxygen mass transfer and shear stress considerations. The bioreactor fluidics was optimized to include a two-pump system, together with an inline filter that enables the maintenance of a constant rate of perfusion through the tissue formed in the channels of the silicon scaffold in the bioreactor. 'lechnology Institute of NLsachusetts Institute ,%assachusetts of'lechnolog _··_ " -E _ ^. U- .- 68 -68 - - Biotechnology Process Engineering center Biotechnology Process Engineering Center -· Chapter 3 Modeling the Fluid Dynamics and Mass Transfer Effects in the 3D Bioreactor This chapter details the fluid dynamical modeling of flow in the microfabricated bioreactor in which primary rat liver tissue is maintained in 3D channels in the silicon scaffold, by continuous perfusion of culture medium. The fluid dynamical modeling of the bioreactor system was done with considerable support from Mohammad Kaazempur-Mofrad, a research scientist with the Kamm Lab. Figures in Sections 3.1 and 3.2 have been taken from already published modeling results [89]. In addition, to better understand the tissue disposition of xenobiotics, nutrients as well as oxygen, a comprehensive mass transfer model was also developed. Perfusion through the channels causes the cells to be constantly under the influence of fluid flow. The rate of flow can influence morphogenesis and cellular function via mechanical and chemical means [81, 84]. Fluid shear can activate signaling pathways and influence differentiated function of hepatocytes and endothelial cells [82, 83]. An accurate determination of the shear stress experienced by the tissue is important in determining several design parameters such as flow rate and geometry to ensure that the cells in the bio-reactor are subjected to the same kind of environment as they would inside the liver capillaries. Fluid flow inside the bio-reactor was analyzed using a computational model in ADINA ® to estimate the velocity profiles, pressures and fluid shear stresses present within the system. The reactor geometry is sufficiently complicated that simple analytical calculations had to be supplemented by a full computational model. In addition to shear stress, the fluid flow through the channels influences the metabolite and nutrient concentration distribution both within the channel as well as in the three-dimensional tissue. These two concentration gradients are coupled and necessitated the use of a Finite Element Model in FEMLAB, to solve for the gradients. Technology Massachusetts Massachusetts Institute Institute of ofTechnology - 69 69- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center The computational fluid dynamical model in ADINA®, together with the mass transfer model in FEMLAB®, allow for the determination of tissue distribution of any metabolite, given an appropriate intrinsiccellular uptake/ release rate for the metabolite. Alternatively, the mass transfer model helps estimate the intrinsicrate of metabolismof any metabolite, given the average outlet concentration of the metabolite in a single-pass of the metabolite through the reactor at steady-state. For a reactor operated in complete recycle mode, the model may still be used to predict the intrinsicrate of metabolism,given the average outlet concentration of the metabolite as a function of time, and ensuring that the dead volume in the reactor fluidics is very small to ensure an average residence time much smaller than the time between consecutive media samples taken from the reactor. This can ensure quick equilibration of local concentration changes within the reactor volume. 3.1 Computational fluid flow model in ADINA The main results reported in sections 3.1 and 3.2 (upto Figure 3.8), was work done by Dr.Mohammad IKaazempur Mofrad and Dr.Arpita Upadhyaya of the Kamm lab, published previously [89]1.The fluid dynamics within the reactor was modeled using the complete form of the Navier-Stokes equations in three dimensions, without any initial relaxations or approximations. The basic governing equations for viscous incompressible fluid flow are (using index notation and usual summation convention): Continuity: Viji= (3-1) 4 i+ vjvi,j Momentum: = Zj (3-2) where p is the mass density of the fluid, ,i is a component of the fluid velocity and ,yis the ij1 component of the stress tensor. Zr,=-pg Inttt Masahset ofTeholg Massachusetts Institute of Technology + 2,e 70 -70 - (3-3) tcnlg : cesEgneigCne Biotechnology Process Engineering Center .... . - e where p is the fluid pressure, ,. is the V. +v.i 2 2 (3-4) ronecker delta, and ,U the coefficient of viscositv. The finite element method was used to solve the above equations incrementally in time using the commercially available finite element analysis software ADINA, version 7.3. The main model developed for the Millill reactor and published previously [89] was slightly modified with the help of Dr.IKaazempur-Mofrad and used for the MilliF reactor. The reactor geometry in the model was made identical to the geometry of the bioreactor used in the experiment. The reactor parts included in the model were - the upper chamber with its inlet and outlet channels as well as the silicon scaffold with the tissue filled micro-channels. The entire volume under consideration was meshed using four-node tetrahedral fluid elements. A linear interpolation function for the velocity and pressure were used with the NewtonRaphson iteration scheme. For the whole-reactor studies, a mesh size of 0.1 mm was used and provided sufficient accuracy. The mesh size was decided as an optimal value based on computational resources and accuracy of the solution. The material properties were assumed to be those of water: density = I g/cm 3 and viscosity = 0.01 Poise. Further, the assumption of laminar flow is justified due to the low Revnolds number of the sstem: Re = 1 in the interior of the upper chamber and Re = 0.1 within the scaffold for finite cross-flow. The value of the experimental input flow rate from the peristaltic pump was used to calculate and specify the input velocity (in the y-direction) at the channel inlet. For simplicity, the average velocity was calculated using v = Q/A, where Q is the pump flow rate and A is the cross-sectional area of the inlet. A flat profile was assumed at the inlet. It was verified that the fluid attains a parabolic velocity profile within a distance of approximately one diameter from the inlet as expected for low Re flows. A zero traction boundary condition was specified at the outlet from the upper chamber of the reactor to denote a free surface. No-slip boundary conditions were specified at the walls of the upper chamber and the silicon scaffold. The outlets from the scaffold micro-channels were either specified with no slip boundanr if Lechnologv Massachuctts M.-asschu sur s IInitinire snsllre off'echnolo gy~ -71-71- Biotechnology Proccs Engineering Centcr BiotechnoloD, Process Elngineering:Center conditions to denote zero cross flow or given a specified velocity in the z-direction (as calculated from experimental values of cross-flow rate). The presence of tissue in the micro-channels was modeled by redefining the channel geometry. Within a day after seeding, the hepatocyte spheroids, which are firmly adherent to the collagen coated channel walls, spread out to form a layverof tissue inside the channels, leaving a roughly cylindrical conduit for fluid flow. The geometry is evident from the SEM images (Figure 3-l). Isometric view "I. 0 El Is 300 1 =. _ I I Figure 3-1: Scanning electron micrographs providing appropriate length scales for the idealized tissue geometry. The isometric view shows the idealized tissue geometry-an annular tissue area with a cylindrical conduit in the center of the channel. Through the obsenrvationof several images (at least 15) of tissue structure in scaffolds, the model geometry of the flow path was defined to be a cylinder of 100 im diameter. The surface was assumed to be rigid with no slip between the fluid and tissue. This assumption is justified considering that the small fluid velocities in the reactor are not sufficient to cause flow induced deformation of the tissue. Two regions can be specifically identified in the idealized geometry in Figure 3-2 - the conduit that allows cross-flow through the channel and the tissue. As a first pass, the tissue was assumed non-porous ind convective transport through the tissue was neglected. A detailed investigation of the stresses inside a single micro-channel required the use of a finer mesh than that used globally. Due to computational limitations, a smaller mesh size of 0.01 mm for only one channel at several scaffold locations was used, while using Technology Massachusetts Maachusetts Insdtute Instituteof offechnolop, _1_ · ·I__ 72 - 72 - - Biotechnology Process Engineering Center Biotechnolop- Process En~ineering Center __ - ~ ~ ~ ~ _ the original mesh of 0.1 mm for the rest of the reactor. Figure 3-3 shows the mesh for an entire model. Isometric view Trj~f "ALILRu Fluid flow E E o0 M Ti Tirr_ Fluid flow path Figure 3-2: Idealized tissue-channel geometrn with a cylindrical conduit for convective flow A non-porous tissue, with species diffusion and enzymatic reaction as the only contributing factors to mass conservation within the tissue, was assumed. p rfuson cross-fow mlcrochannels Figure 3-3: Mesh distribution in the ADINA ® model used to describe fluid flow in the bioreactor (from [891) Inttt of1cn-g'-3 ~\Lssahustt ManssachusettsInstitute of Te~chnolop- 73 itcnloyPoesEgneigCne Biotechnolop, Process Einceri~ Centcr 3.2 Velocity profiles and shear stress distributions This section details the results from the fluid dynamic modeling of flow in the bioreactor. The input flow rate from the pump was set to be 0.5 ml/min and the rate of crossflow was specified to be 40 .d/min (or 1 l/min per channel) based on experimental data. Figure 3-3 shows the y-velocity (along the long axis of the chamber) at the vertical center plane (z = 0.0). No apparent non-uniformity of flow is seen along the length of the reactor. Figure 3-4 shows the x component of the velocity at the center z-plane. The xvelocities are high near the inlet and outlet due to the sudden widening and narrowing of the flow path, but reduce to zero beyond at most one row of channels. Therefore an axial flow is maintained over most of the channels. The z-velocities are negligibly small at this plane. Z X Y Y-VELOCITY TIME1.000 - 1E.80 )W - 15.60 - 1440 - 1a20 ' N., - 10.80 .1~ - % -~:., -- 1 ..> - 9.60 8.40 7.20 6.D0 4.80 3.60 2.40 1.20 00 Figure 3-4: Uniform flow seen over the top of the channels, over most part of the bioreactor after a short entrance length (from [89]) Inttt tehoov-7 Mnsahset Nhisgsachusett, nstitute of Technology ..... .- -74 - itchooyPoe EgneigCnc BiotchnologyS Process Engineering Center 5~~~~~~~~~~~~~~~~~~~~_. A- b z Y-VELOCITY TIME1.000 L[~~I- i'Ag~~ O W 14.40 13.20 -12.0U -10.80 9.60 8.40 720 6.00 4.80 3.60 2.40 - 1.20 0.00 Figure 3-5: Uniform axial flow is seen to be maintained over most of the channels (from [891) It is seen that the flow is fully developed away from the inlet and outlet. Next, the effects of fluid flow inside single tissue-filled microchannels within the silicon scaffold were investigated. The main effect of shear stress will be on the outermost cell layer in contact with the fluid. The cells lining the cross-flow path are directly under shear and therefore susceptible to shear induced mechanical and chemical signals. The experimentally obtained value of roughly 1 l/min per channel (experimentally determined to be in the optimal range wherein CYP450 expression is seen not be affected by flow rate) was used to specify the z-velocity at the channel outlet. Figure 3-5 shows the z-velocity inside a single channel of 100 Jim diameter at the center of the scaffold. The flow becomes fully developed within 50 Sim of entering the channel. Figure 3-6 plots the wall shear stresses at different z-positions along the tissue filled channel. The shear stresses at the walls are about 0.4 dyne/cm 2 near the top of the channel and approximately 0.7 dyne/cm 2 halfway through the channel vertically. To verify these results, the wall shear stress in the channels was calculated assuming Poisseulle flow. Using the relation: ;' = 8luQ,/2 3, z' = 0.7 dyne/cm 2 , similar to the value obtained from the numerical Technology of Technology Institute of Massachusetts Institute Nfaachusetts -75- -75 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center ·_ model. Figure 3-7, shows a pretty uniform shear stress distribution over the top of all the channels in the bioreactor. L z x. Lv VELOCITY Z-VELOCITY TIME 1 000 TIn 1.000 3500 - - -00 O ' 2.750 --.000 -1.250 -1 W -1.7500 -2.?.. - 2.000 -22W -1.000 2.750 -0.750 0.500 -2.750 a' :. -3.250 -3.500 i 0.250 0.000 Figure 3-6: Velocity profile in a single channel of the bioreactor (from [891) The presence of the filter causes the profile to flatten at the end of the channel. Figure 3-7: Shear stress distribution along the depth of a channel ('s' denotes distance from the top of a channel). The maximum shear stress experienced by the cells is on the order of 0.07 Pa; well within the range of physiological shear stress values (from [89]). Massachusetts Institute of Technology - 76- Biotechnology Process Engineering Center Perfusion flow: 40 ul/min i ' Axial flow: 500 ul/min ss'~ 0.030 0020 _ 0.015 0.010 o. n" )0O 8x3 MilliReactor with open cross-flow channels Figure 3-8: Uniform shear stress distribution seen over the top of the channels in the bioreactor (from [89]) In order to determine the appropriate range of crossflow rates that simulate physiologically relevant shear stress rates to enable the histotypical reorganization of the 3D tissue, the maximum shear stress experienced by the tissue in a channel was plotted as a function of the crossflow rate. This has been reproduced in Figure 3-9. The Poiseullar nature of the flow predicted by previous modeling studies [89], was thus confirmed using the full blown ADINA model developed for the MilliF bioreactor by Dr.Mofrad. Based on the range of flow rates seen in in vivohepatic sinusoids[81, 83, 84, 93], the perfusion crossflow rate may be set to a value between 40 l/min and 600 l/min, to mimic shear rates seen in the in ivo microenvironment. Technology of Technology Institute of Massachusetts Institute Massachusetts - 77 77- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center II 0.25 W0.20 c0.15 co Physiological Value - lower bound for )O perfusion cross-flow rate, uL/min Figure 3-9: Maximum shear experienced by the cells in a channel as a function of crossflow rate. Based on reported in vivovalues of shear experienced by cells in the hepatic sinusoids, the bioreactor may be operated in a range of flow rates between 40 ul/min and 0.6 ml/mnin. 3.3 Mass transfer model in FEMLAB® An idealized tissue-channel geometry with a cylindrical conduit for convective flow, as seen in Figure 3-2 formed the basis for the mass-transfer model. A non-porous tissue, with species diffusion and enzymatic reaction as the only contributing factors to mass conservation within the tissue, was assumed. The shaded area in Figure 3-2 represents the tissue. The basic species conservation equations were used to solve for the coupled tissue-conduit species concentration profile. These equations in their most generalized form are (assuming constant density and diffusivity): DC = DV 2 Ci + R Dt (3-5) ~- Inttt fTcnlg ~ ~ ~~ ~ -787-BoehooyPoesEgneigCne Massachusetts~~ Massachusets Institute of echnology Biotechnology Process Engineering Center +, Where: a a D -- v +-- a +v (3- a - and D i is diffusivity of the i species, C i is the concentration of the i species in the tissue, R vi , is the cellular uptake or release rate of the ih species, r, 0 and z represent the cylindrical coordinates used to describe the system. Since the tissue is assumed solid and non-porous, the species conservation equation at steady state applicable to the tissue (shaded area in Figure 32), reduces to: DiV2Citi,ssie + R,, = 0 (3-7) Where Citissue represents the i h metabolite concentration in the tissue. The appropriate boundary conditions needed to solve for the concentration profile in the tissue include the noflux boundary condition at the walls of the chip, flux-continuity at the tissue-fluid interface, and appropriate concentrations on the top and bottom plane surfaces of the scaffold (see Figure 3-7). - D.VC, isse - D.VCi,tiue Mwall =0 (3-8) D.VCi,fluid fluid-tissue tissue-fluid= (3-9) where CO, and Cz=r are set to known inlet substrate concentrations Co. The species conservation equation in the channel with the Poiseuille flow approximation (which follows from the fluid dynamic modeling described in the previous section) reduces at steady state to: Techno1o Massachusetts Instute Massachusetts Institute of ofTechnology - 79 - -79 - Biotechnology Process Engineering Center Biotechnology Process Engineering: Center -v~pi = D.V2Cibflid z (3-10) z The appropriate boundary conditions needed to simulate the metabolite distribution in the channel include the continuity of fluxes at the fluid-tissue interface and a valid inlet concentration of the metabolite. - D.VCi,fluid fluid-tissue= Cfluidz=T D.VCissue (3-11) tissue-fluid (3-12) = Co The species conservation equations were non-dimensionalized using an appropriate length scale (a fixed value of 100 m, which is in the range of the diameter of the cylindrical flow conduit seen, L), an appropriate concentration scale (inlet metabolite concentration C), and an appropriate velocity scale (U=vzinet is the velocity entering the channel at the bottom of the scaffold). This leads to the description of the system using the following nondimensional parameters: The PNcletnumber (Pe),and the Damkohler number (Da) or Thiele modulus (). Convectlon-dominated flow on top of the scaffold - Longitudinalaxis of symmetry -z Z=O ~ 'L : Tissue - fluid Interface .i . *. Z=T -- flowthroughchannel Convectlon-dominated (P6- 100 for a cross-flow rateof 1 plVmln/channel) L Figure 3-10: Interfaces and boundaries in the mass transfer model Massachusetts Institute of Technology - 80- Biotechnology Process Engineering Center The appropriate experimental values of inlet velocity U=vi,,le, , inlet substrate concentration C,, and the characteristic bioreactor channel dimensions (square cross section of 300 munand a depth of 230 Etm, characteristic length scale L = 100 pm) were used to determine the values of the non-dimensional parameters C*, Pe, and Da defined in Figure 3-8. Time scalefor convectivetransport *Pclet · Number: Timescalefor diffusive transport UL D Time scalefor reaction ·Thiele Modulus/ Damkohlcr Number: R.L2 Time scale for diffusive DC transport through the tissue Cross-section P ~ 1(3 Fluid -tissue *= 1 cell - interface P varies with cross-flowrate Figure 3-11: Appropriate non-dimensional parameters used to characterize the system The average viable cell number in the tissue in a channel was determined from total RNA measurements, described in the Appendix. The planes of symmetry seen allowed the reduction of the FEMLAB® simulation to the modeling of a quadrant of the tissue-channel geometry. The cell uptake or release rate of a metabolite was assumed to follow MichaelisMenten kinetics. It is defined as: RV =Vmax C C+Km Technology Instute of Massachusetts Massachusetts Institute ofTechnology - 81 - - 81- (3-13) Biotechnology Process Engineering Center Biotechnology Process Engineering Center where V, and K, are the appropriate Michaelis-Menten parameters, representing the maximal enzyme activity and the substrate concentration at which the activity is half the maximal, respectively. Tetrahedral elements were used to set the mesh distribution in the geometry. Mesh sensitivity analysis was performed at the end of every simulation to check for the effect of mesh density of the predicted concentration profiles. Non-dimensionalization of the governing species conservation equations yields two main non-dimensional parameters RL2 , which represents the ratio of the time scale for species the Damkohler number (~' DC, reaction to the time scale for species diffusion to the site of reaction in the tissue), and the UL which represents the time scale for diffusive to convective transport of Pclet number (-, species through the cylindrical fluid conduit). In the definitions above, L is the characteristic length scale (taken to be 100 pm), D is the species tissue diffusivity (m2 /sec), U is the average Poissueller velocity (m/sec.), C,, is the initial species concentration at time t = 0, and R,.is the volumetric species consumption or production rate (also called the volumetric reaction rate, moles/m 3 sec.). 3.4 Tissue disposition of oxygen The model was used to simulate the tissue and channel distribution of oxygen using parametric values reported in literature. A maximal, conservative (i.e. the highest value reported in literature) cellular oxygen consumption rate of V,/ = 0.4 nmoles/ 106 cells/ sec. [94], an inlet oxygen saturation concentration of 0.19 mol/m 3 (corresponding to a volumetric ratio of 19.2% 02, 72.3% N2 and 8.5% CO), a KM of 7 M [94], and an oxygen tissue diffusion coefficient of 1.9 x 10-9 m2 /sec. [95, 96] were used as parameters in the model. The per cell consumption rate expressed in units of nmoles/10 6 cells/sec. was converted into a volumetric rate, expressed in units of nmoles/ m3 of tissue/ sec. using a calculated value of tissue volumetric density expressed in units of cells/m3 (Table 3-1). Based on the underlying assumption made, two different scenarios exist for the tissue disposition of oxygen: Technology Institute of Massachusetts Institute Massachusetts ofTechnology - 82 82- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center 1. The cells are rigid and each cell occupies a finite constant volume in a tissue, so that the radius of the cylindrical conduit in Figure 3-2, changes with the average cell number per channel. 2. The cells are flexible and can be packed in a finite fixed tissue volume - i.e. the mechanical and biophysical forces force a cylindrical conduit of a fixed radius in each channel independent of the cell number. Transmission electron micrgraphs such as those in Figure 4-7, indicate that in some cases the cells are non-spherical and compacted in a tissue, while in some other cases they maintain their rounded morphology. Thus both scenarios are analyzed for the tissue distribution of oxygen. For the first scenario, the tissue volumetric density (cells/m3 ) was calculated using the theoretical maximum number of spherical cells of 25 rmndiameter that may be packed in a total channel cuboidal volume of 300 upmx 300 pm x 230 Pm) and is equal to 1/VC = 1 /(4/3..r3), where 'r' is the radius of a single spherical cell (12.5 Vm).The average cell number per channel is calculated using the value of total cell number obtained by measuring the total RNA (see Appendix) in the scaffold in the bioreactor. It should be borne in mind that though there may be variations in the total cell number per channel across different bioreactors - this does not affect the cell density (cells/m3), but does affect the effective diameter of the porous conduit available for perfusion flow. Hence while the Pe number varies as a result of the differences in cell number (causing variations in the effective cylindrical conduit radius available for flow), the Damkohler number remains the same. Table 3-1 describes this in greater detail. In order to study the effect of various perfusion or crossflow rates on the tissue distribution of oxygen, experiments were performed with a set of eight reactors set at different flow rates - 0, 40, 100, 230 ul/min (two reactors at each setting). Day 3 spheroids were seeded into the reactors and cultured upto seven days in culture post cell-isolation. Table 3-1 gives the cell numbers measured in the bioreactors and the calculated values of the Pe and Da numbers. Technology ofTechnology Institute of MassachusettsInstitute 83 -83 - - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Table 3-1. Average viable cell numbers and non-dimensional parameters for modeling the tissue distribution of oxygen in bioreactors with and without cross-flow - Scenario 1 Bioreactor Avg. viable cell Avg. viable cell Calculated Effective cross-flow (ul/min) number based on total RNA number per channel Tissue Volume, using Tissue length scale 't' (prm) measurements 0 27275 +/- 6909 682 a cell diameter of 25 !zm [m 3] 5.582 x 10-12 40 38956 +/- 9677 974 7.972 x 10-12 18 100 25111 +/- 5620 628 5.140 x 10-12 3.5 230 32527 +/- 3250 813 6.654 x 10- 12 11 Diameter of cylindrical crossflow Perfusion velocity per channel (m/s) Characteristic Length Scale used for nondimensionalization Peclet Number (Pe) Damkohler number (Da) 100 0.00 1.35 (n=4) conduit (300 - 2*t) 290 5 (Pm) 0 264 1.9022E-05 100 1.00 1.35 293 3.86073E-05 100 2.03 1.35 278 9.86376E-05 100 5.19 1.35 Figure 3-12 shows the predicted tissue distribution of oxygen for the case where there is no crossflow (using parameters from Table 3-1). A non-dimensionalized concentration of 1.0 corresponds to saturated concentration of oxygen in the media in equilibrium with air containing 19.2% of 02, 79.3% N2, and 8.5% CO 2. The oxygen concentrations drop almost to values comparable to IM at the bottom (230 Gm) of the channel However, tissue hypoxic responses at the gene expression level can occur at low but non-zero oxygen concentrations (7% 02 or 0.36 in the scale where 1.0 corresponds to dissolved oxygen concentration in the medium in equilibrium with 19.2% 02, 72.3% N2 , and 8.5% CO) [97]. Such concentrations are reached at a depth of 130 pm from the top of the channel or 100 plmfrom the bottom of the channel. Thus, a large volume of tissue (nearly 43%) in the bioreactors with zero crossflow is exposed to hypoxic conditions. In contrast, in bioreactors with a cross-flow rates of Technology Massachusetts ,Massachusetts Institute Institute of ofTechnology - 8484- Biotechnology Process Engineering Center Biotechnology Process Engineering Center even 40 pl/min (i.e lpl/min per individual channel), the minimum oxygen concentration in the tissue (0.57) is significantly higher (Figure 3-13) than concentrations at which tissue hypoxia effects have been reported in literature (0.36). Max 1 1 0.9 250 0.8 200 0.7 150 0.6 100 0.5 50 0.4 0.3 0 0.2 -50 0.1 I 150 (a) 150 (b) u Mi 0.0473 0 Figure 3-12. Tissue distribution of oxygen in the limiting case of reactors with zero cross-flow - Scenario 1 The oxygen concentrations are predicted to drop to a value lower than 0.36 at a depth of 130 pumfrom the top of the channel especially in the cells located right at next to the walls of the channel (a) A large volume of tissue (nearly 43%) in the zero cross-flow reactors is exposed to hypoxic conditions Figure 3-12(b) shows the oxygen distribution in the cylindrical porous conduit. (Note: Symmetry reduces the problem to solving the concentration profile in onequarter of the channel and tissue occupying volume of 150 x 150 x 230 pn). - 85 MassachusettsInstitute of Technology - 85Massachusetts Institulte of Technology Biotechnology Process Engineering Center~~~~~~~~~~~~~~~~ Biotechnology Process Engineering Center Max:1 250 0.95 200 0.9 150 0.85 250 200 150 0.8 100 100 0.75 50 0.7 50 0 0.65 0 -50 -50 0.6 15 150 150 150 u Mio.565 0 Figure 3-13: Tissue distribution of oxygen in bioreactors with cross-flow - Scenario 1 In reactors with a crossflow rate as low as 40 pl/min, the minimum oxygen concentration in the tissue, even at the walls of the channel farthest from the flow eft side figure) is predicted to be significantly higher (0.57) than concentrations at which tissue hypoxia effects have been reported in literature (0.36). The figure on the right hand side shows minimal drop in concentration in the cylindrical conduit (Note: Symmetry reduces the problem to solving the concentration profile in one-quarter of the channel and tissue) For the second scenario, the tissue volume was assumed constant independent of the cell number (i.e. the radius of the flow conduit, was assumed constant and equal to 100 im). The volumetric density (cells/m3 ) was calculated using the experimentally measured number of cells. The average cell number per channel is calculated using the value of total cell number obtained by measuring the total RNA (see Appendix) in the scaffold in the bioreactor. It should be borne in mind that though there may be variations in the total cell number per channel across different bioreactors - this now affects the cell density (cells/m3), but does not affect the effective diameter of the porous conduit available for perfusion flow. Hence while the Pe number remains constant even with differences in cell number, for the same crossflow flow rate, the Damkohler number changes with changes in tissue volumetric density. A characteristic length scale equal to the assumed tissue thickness (or of the cylindrical pore Massachusetts Institute of Technology -86 - Biotechnology Process Engineering Center conduit diameter) of 100ptm, is used in this scenario. Table 3-2 describes this in greater detail. Table 3-2 gives the cell numbers measured in the bioreactors and the calculated values of the Pe and Da numbers for Scenario 2. Table 3-2. Average viable cell numbers and non-dimensional parameters for modeling the tissue distribution of oxygen in bioreactors with and without cross-flow - Scenario 2 Bioreactor Avg. viable cell (A) (B) Tissue Effective cross-flow (ul/min) number based on total RNA Avg. viable cell number per Fixed Tissue Volume [m3] Volumetric Density Tissue length [cells/m3] = scale 't' A/B (gm) measurements channel (n=4) 0 27275 +/- 6909 682 1.889 x 10-11 1 3.610 x 1013 100 40 38956 +/- 9677 974 1.889 x 10-" 5.156 x 1013 100 100 25111 +/- 5620 628 1.889 x 10-1 3.325 x 1013 100 230 32527 +/- 3250 813 1.889 x 10-11 4.304 x 1013 100 Diameter of cylindrical crossflow conduit (300 - 2*t) Perfusion velocity per channel (m/s) Characteristic Length Scale used for nondimensionalization (pmr) Peclet Number (Pe) Damkohler number (Da) 100 0 100 2.121 x 10- 100 100 100 0.00 0.400 3 100 112 0.571 3 100 280 0.368 100 644 0.477 5.303 x 10- 12.197 x 10- 3 Note: In this scenario, the tissue volume in the bioreactor was assumed constant independent of the number of cells. i.e. the volume corresponded to the space between the cuboidal channel volume (230 }m x 300 ptm x 300 tm) and the cylindrical annulus seen in Figure 3-2 (of diameter 100 u[m).Thus, it was assumed that the cell-packing density changed to accommodate more cells within a given volume.. The conclusions drawn from 3-14 and 3-15 are exactly the same as the conclusions drawn from the predicted tissue distribution of oxygen in Figures 3-12 and 3-13. Thus, both scenarios lead us to the same conclusion - in the absence of crossflow, a large part of the tissue is exposed to hypoxic conditions, while any crossflow rate greater than 40 ul/min Massachusetts Institute of Technology -87 - Biotechnology Process Engineering Center through the forty channels of the bioreactor, helps satisfy the tissue oxygen demand and helps maintain normoxic conditions in the tissue. .. . It n 0.0 E 100. a. ..' 50 "'i _j 0 1 . .. 0 150 .. 200, 0.9 Z 0.8 ' " 0 : .:" 250 L, ,... 0/ 100 50 9',- so . ' ... . .. .::: ,..~.O .. .. . .... ··':':' -""" 150 O 0.2 0.1 :. 100 Rol 0 50 0.5 0.3 ' 150 0.6 L..0.4 200 250, 150 z 0.7 w o 150. z Max:1 . 100 . .... 50, .: Z : _01i" -. .z- u50,. 150 LOO lt V~ f4 Figure 3-14. Tissue distribution of oxygen in the limiting case of reactors with zero crossflow: Scenario 2: The oxygen concentrations drop zero at a depth of 130 pm, as can be seen in the plane cross sections of the channel, in the first figure. A large volume of tissue (nearly 43%) in the zero cross-flow reactors is exposed to hypoxic conditions (Note: Symmetry reduces the problem to solving the concentration profile in one-quarter of the channel and tissue occupying volume of 150 x 150 x 230 pm). Massachusetts Institute of Technology - 88- Biotechnology Process Engineering Center m.I a: UI 9 u.U F1 0 50 Cr A A - 1200 150 . oz 0 0.9 MI 0.0 z ~95 1 50 0 81 0.95 uJ 200 0 < 075 250 : O 07 0.75 O 0.6089 u1515 0 ' 50 0 MA0.7M 150-150 0 50 a) Cross-Flow Rate: 401uUmin 50 b) Cross-Flow Rate: 230 l/min Figure 3-15: Tissue distribution of oxygen in bioreactors with cross-flow - Scenario 2. In reactors with cross-flow rates in excess of 40 A/min,the minimum oxygen concentration in the tissue (0.69-0.74) is significantly higher than concentrations at which tissue hypoxia effects have been reported in literature (0.36). (Note: Symmetry reduces the problem to solving the concentration profile in one-quarter of the channel and tissue). High Pclet numbers, seen for Scenario 2, result in convection-dominated profile through the porous non-tissue conduit, which in turn translates to a negligibly small concentration boundary at the tissue-fluid interface. Thus, the minimum tissue concentration of oxygen becomes independent of Pclet numbers at values of Pe > 100. This results in all the minimum concentration-Damkohler number curves in Figure 5-18 to collapse into a single curve, at Pe>100. Similarly, at very low P6clet numbers (<0.1), the minimum tissue oxygen concentration once again becomes independent of Pdclet number, as can be seen in Figure 316. Massachusetts Mvassachusetts Institute Institute of of Technology Technology; 89 -89 - - Biotechnology Process Engineering Center Biotechnology Process Engineerinig Center Minimum Tissue Oxygen Concentration as a function of the Damkohler Number (Thiele Modulus) an 1.u 0.9 _ 0.8 = * ' 0.7 0.6 ' C 0.5 E C 0.4 0.3 a 0.2 0.1 0.0 0.00 0.50 1.00 1.50 2.00 2.50 3.00 3.50 4.00 4.50 Damkohler Number (Da) Figure 3-16: Minimum tissue oxygen concentration asymptotically falls to zero for large values of the Damkohler number (Da). Hypoxia-like conditions set in only at values of Da > 1.4 in the bioreactors with cross-flows greater than 40 l.d/min(Pe= 112). 3.5 Measurement of the effect of crossflow In order to validate the model mass transfer predictions within a tissue containing channel, expression of hypoxia regulated genes Hif-3ot and Heme Oxygenase -1 were studied as a function of the cross-flow rate (see Fig. 3-12 and 3-13, respectively). Both Hif-3a and Heme Oxygenase-1 have been well studied in literature as hypoxia-responsive genes [97, 98]. The object of the gene expression analysis was to determine an appropriate range of crossflow rates at which tissue oxygen concentration profiles are well above those that induce tissue hypoxic responses. The range of perfusion or cross-flow rates at which the shear stress experienced by the cells in the tissue are physiologically relevant, while not being too high as to adversely affect the tissue phenotype (determined by looking at the expression of CYP3A as a Inttt fTcnlg Mascuet Massachusetts Institute of Technology - -90 - Bitcnlg Prcs Eniern Cete Biotechnology Process Engineering Center function of the crossflow rate - see Figure 3-17), were used to study the effect of perfusion crossflow rate on oxygen distribution in the tissue. I I 22 40 40 75 105 230 105 230 Flow rates in ul/min Figure 3-17: Effect of perfusion crossflow rates on tissue expression of CYP3A2. Perfusion crossflow rates upto 230 ul/min are seen not to affect the expression of p450's. All lanes normalized to total protein Relative to the expression in isolated hepatocytes and the bioreactors with cross-flow, the expression of Hif-3-c mRNA in the bioreactors without cross-flow is significantly higher (Students t-test, p<0.05, Figure 3-18). The mass transfer model had shown the presence of hypoxia-like conditions in a significant volume of tissue in the bioreactor with zero cross-flow, relative to tissue structures in bioreactors with cross-flow (Figures 3-12 to 3-15). We believe this may be responsible for the induction of Hif-3c in reactors without cross-flow (Figure 316). Induction of Hif-3 ocin response to mild hypoxia has been reported previously [97], although the study had concluded that the induction of Hif-3c in in ivo experiments on rat liver was not very significant. However, the duration of exposure to mild hypoxia in the studies (up to two hours) was significantly shorter than in our bioreactor experiments (nearly 72 hrs.). Heme Oxygenase -1, which has been shown to be induced in response to mild hypoxia [98], was induced nearly three-fold in the bioreactors without cross-flow relative to those in non-zero cross-flow reactors (Figure 3-19). These results confirm the model Technology Massachusetts Institute Massachusetts Institute of ofTechnology -91-91 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center prediction of hypoxia-like conditions in the bioreactors without cross-flow. This study did not find Hif-la to be very responsive to hypoxia (datanotshown)at the mRNA level. T -I1 5.00 4.00 U' C 2. 2.00 2.00 freshly isolated T C IL 1.00 @ .00 · .2 0.00 @5 -1.00 1. -2.00 I I.................................................................................. Perfusion cross-flow rates through 40 channels rul/minl Figure 3-18: Expression of Hif-3-c mRNA in bioreactor cultures operated at different crossflow rates using RT-PCR Induction of Hif-3-alpha expression relative to basal levels can be observed in reactors without cross-flow (marked by *). The induction is statistically significant (at the 95% confidence level) in zero cross-flow bioreactors, compared to the expression levels of Hif-3-ocin the bioreactors with non-zero cross-flow rates. Results are from N=2 experiments with n = 2 bioreactors at each flow setting in each experiment. In the bioreactors with cross-flow in excess of 40 l1/min(Pe = 112, for Scenario 2), the model predicts -onset of hypoxia-like conditions (defined by oxygen concentrations below 0.36) at Damkohler numbers greater than 1.5. However, in all experiments in bioreactors with cross-flow, Damkohler numbers were in the range of 0.40-0.62 for Scenario 2- values much lower than 1.5. Tissue oxygen concentration asymptotically falls to zero for large values of the Technology of Technology Institute of Massachusetts Institute Massachusetts 92 -92 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Damkohler number (Da). (Figure 3-16). The intent of the RT-PCR studies was to identify suitable range of cross-flow rates at which no hypoxia was observed in the channels of the scaffolds. This was seen to be the case for all cross-flow rates in excess of 40 pl./min. Nonetheless, die presence of non-parenchymal cells, such as the activated Kupffer cells, may lead to an increase in oxygen uptake rate [99, 100] and hypoxia-like conditions. In these experiments, Percoll centrifugation during cell isolation minimizes the concentration of nonparenchymal cells in culture. _ , I . 4.U 0 3.00 o ca la 2.00 (3 1.00 co O o,.. 0.00 -1.00 -00 -P fl Figure 3-19: Induction of Heme Oxygenase - 1 mRNA in bioreactor culture under hypoxia: Heme Oxygenase - 1 is induced in reactors without cross-flow. This induction is statistically significant (at the 95% confidence level, p < 0.04), compared to the expression levels of Heme Oxygenase - 1 in the bioreactors with non-zero cross-flow rates. Data in (A) is from RT-PCR studies. Results are from N =2 experiments from 2 different rats and n = 2 replicate reactors at each flow rate in each experiment. Expressions in all bioreactors run with cross-flow at 40, 100 and 230 pl/min were comparable (data not shown). Data in (B) is from RatU34A microarray expression data (seeChapter 5for details on basalgene expressionstudies) 3.6 Summary of key results and conclusions The computational fluid mechanical model in ADINA ® was used to describe the velocity and shear stress distribution within the bioreactor. The model helped identify a set of Technology Institute of Massachusetts Institute Massachusetts ofTechnologyr -93 -93 -- Biotechnology Process Engineering Center Biotechnology Process Engineering Center crossflow (perfusion) flow rates that simulate physiological shear stress conditions at the tissue surface within a channel of the bioreactor. The mass transfer model in FEMLAB® was used to describe the tissue distribution of metabolite concentration gradients. The mass transfer model helped identify appropriate bioreactor operating conditions (Peclet and Damkohler numbers) under which the tissue oxygen demand was theoretically and experimentally validated to be satisfied. The mass transfer model may also be used to determine the tissue disposition of any metabolite, so long as the requisite model parameters - the Peclet Number and the Thiele Modulus or the Damkohler number are known. Massachusetts Massachusetts Institute Institute of of Technology Technology 94 -94 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Chapter 4 Basic Characterizationof Liver Tissue Phenotype in the Bioreactor Enriched rat liver hepatocytes were isolated from male Fischer F344 rats (Charles River, Cambridge, MA), pre-aggregated into spheroids in spinner flask cultures and then seeded into bioreactors. This chapter details the steps involved in the isolation, preaggregation, and culture of the primary rat hepatocytes. A schematic of the steps involved is presented in Figure 4-1. Separate sub-sections in this chapter; give a detailed analysis of each individual step. Cell Culture Methods : Isolation-Seeding Static cultures , ..... l Iaay uj)r (day 3) Spinner flask A A (control) Bioreactor seedina ds) ( Morphogenesis Within 1-2 days. Hepatic functions Isolation: Hepatocyte fraction contains -5% non-parenchymalcells monitored Albumin, Urea, CYP450 expression and activity In vivo liver section Figure 4-1: Schematic of the steps involved between the isolation and seeding of rat liver cells into the bioreactor Massachusetts Institute Massachusetts Institute of of Technology Technology -95-95 Biotechnology Process Engineering Center Biotechnology Process Engineering Center 4.1 Isolation of primary rat hepatocytes A schematic of the process of isolation of enriched rat liver hepatocytes is presented in Figure 4-2. Hepatocytes were isolated from 150-230 g male Fischer rats with a modification of Seglen's two-step collagenase perfusion procedure [101] as described previously. 2-step collagenase I Perfusion (Seglen et al) Cell isolatic)n l- t NPC's &W 1. Intact Liver Tissue 2. Perfused Liver Tissue 3. Non-Parenchymal Cells NPC's) + Hepatocytes soup r l Add percol 140% (v/v) 5-10 million cells/ mL Centr ifuge 3 times cP50g, 3 min. Dead cells, NPC's Centrifuge, 50g 10 min. Nearly 99% pure hepatocyte fraction I 3. Non-Parenchymal Cells (NPC's) Hepatocytes. soup 4. Hepatocyte enriched fraction (95% hepatocytes, J 5% NPC's, Seglen etal) Centrifuge 3 times @509 I 5.Hepatocyte enriched fraction (99% hepatocytes) Figure 4-2 A schematic of enriched rat liver hepatocyte isolation procedure The resulting cell suspension was centrifuged three consecutive times at 50g (2 min each). Next, 21.6 ml of Percoll (Sigma-Aldrich, St. Louis, MO), was mixed with 2.4 ml of Hanks Balanced Salt Solution (HBSS, Sigma, MO), and added to 25 ml of the centrifuged cell suspension at a cell density of 5-7 million cells per milliliter of solution, as described in the literature [102]. The solution was further centrifuged at 50g for 10 minutes. Percoll isogradient centrifugation resulted in the separation of dead cells as well as a significant portion of the non-parenchymal cells in a floating top layer that was discarded. The sedimented hepatocytes were then re-suspended in hepatocyte growth medium (HGM) [103], but without hepatocyte growth factor (HGF). An error was reported in the original HGM formulation reported by Massachusetts Massachusetts Institute Institute of of Technology Technology -96 -96 -- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Block et al. [103]. The concentration of Niacinamide, and those of heavy metal salts were incorrectly reported in the original reference. Thus, the media formulation used for all experiments in Chapters 5, 7, and 7 of this thesis used the corrected media formulation. The concentration of nicotinamide or niacinamide was corrected to 0.305 g/L from the original concentration of 0.61 g/L ZnC2 concentration in the final formulation was reduced by tenfold to 0.0544 mg/L. Similarly ZnSO 4*7H 2 0 concentration was reduced ten-fold to 0.075 mg/L. CUSO4*5H2O concentration was corrected to 0.02 mg/L from the original concentration of 0.20 mg/L. Glutamine concentration was reduced to 1 mM from an original concentration of 5 mM. Gentamycin and HEPES were not used in the media, while Penicilli and Streptomycin were used as bacterial antibiotics in the media. Other components in the media were the same as those reported in [47, 89]. The final cell viability, as determined by trypan blue exclusion, was approximately 90-95% at the end of the isolation procedure. 4.2 Formation of multicellular spheroidal aggregates Thirty million percolled primary rat hepatocytes were added to 100 mL of HGM in a 500 mL spinner flask (BellCo, NJ, USA) on a spinner table set at 85 rpm [20]. The cells aggregated to form spheroids. Flasks were taken down either on Day 2, Day 3 or Day 4 after cell seeding, for seeding the spheroids into the bioreactors. Spheroids of the desired size range (100 - 300 pun) [47], were separated using appropriately sized filter meshes (Sefar America, Kansas City, MO) and re-suspended in 25 mL of rinse medium. The rinse solution comprised phenol red-free Dulbecco's modified Eagle's medium (DMEM) with sodium pyruvate (110 mg/mL) and glucose (1 g/L) (Life Technologies, Rockville, MD) supplemented with bovine serum albumin (2 g/L, Sigma, St. Louis, MO) and penicillin-streptomycin (100 U/mL). The size-separated spheroids were centrifuged at 40g for 3 min. The floating debris was then removed and the spheroid pellet was re-suspended in 30 mL of HGM. Previous work in the lab has indicated minimal differences in tissue phenotype, assessed by measurement of albumin and urea secretion rates in reactors seeded with Day 2, Day 3, or Day 4 spheroid seeded reactors 1471]. Massachusetts assachusetts Institute Institute of of Technology Technology 97 -97 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center 4.3 Bioreactor assembly and cell-seeding imaging & spectroscopy vent flilt Figure 4-3: Schematic diagram of the bioreactor fluidic system The design and assembly of the microfabricated bioreactors have been described in detail in Chapter 2, as well as in earlier published literature [47, 89, 104]. The bioreactor system was primed with rinse solution (comprising phenol red-free Dulbecco's modified Eagle's medium (DMEM) with sodium pyruvate - 110 mg/mL and glucose - 1 g/L (Life Technologies, Rockville, MD) supplemented with bovine serum albumin - 2 g/L, (Sigma, St. Louis, MO) and penicillin-streptomycin at 100 U/mL) to passivate the bioreactor, connector and tubing surfaces, and to remove air bubbles from the flow paths. Prior to seeding, small volume conical glass reservoirs were aspirated and refilled with 2 mL of HGM. For hydrodynamic seeding, a syringe filled with 1 ml cell-spheroid suspension was placed at the inlet of the upper chamber (port #1) and a second syringe containing -0.5 ml of PBS was placed at the bioreactor upper outlet (port #2) (see Figure 4-1). The bottom bioreactor outlet (port #3) was unclamped by removing the tubing from the peristaltic pump rotor and the bioreactor was tilted approximately 450 with the bioreactor outlet (port #2) higher than the inlet (port #1). On the third day, spheroids of the desired size range (100 - 300 Lm)were separated from the spinner flask cultures, using appropriately sized filter meshes (Sefar America, Kansas City, MO) and re-suspended in 25 mL of rinse medium. We have previously shown better functional tissue phenotype in reactors seeded with spheroids rather than with AsAc_.n s I T__. .. c gh IvMassacusets insttute of I echnologyr -98 - yn Biotechnology Process Engineering Center individual cells [47]. The cell-spheroid suspension was manually injected into the upper chamber at the flow rate of -0.5 ml/min. The spheroids entered into the scaffold channels through a combination of settling and flow of cell culture medium from the upper chamber into the lower chamber aided by the resistance of the syringe on the bioreactor upper outlet (port #2). Following seeding, cell culture medium was pumped through the upper bioreactor chamber at 0.5 ml/min (with pump A) and collected in a waste container for one minute to clear cells from the top surface of the scaffold. The upper recycle tubing was then reconnected to the bioreactor (port #2). At this time, the bottom tubing was inserted into the peristaltic pump B and the perfusion flow rate (cross-flow) down through the seeded channels was set to a desired flow rate. The selection of flow rates was based mainly on being able to simulate physiological shear stress conditions, while satisfying tissue oxygen demand, in the tissue units in the scaffold channels. The choice of these flow rates has been described in detail in Chapter 3. After one hour, cell culture medium in the reservoir was replaced with 2 ml of fresh medium to reduce residual cells/debris trapped in the circulation loop. Spheroids seeded into the channels are initially held in place by the membrane or filter, and after initial attachment and reorganization (-1 day), by adhesion to the collagen-coated channel walls. During the "top down" perfusion, the residual cell debris is likely to gradually clog the micro porous filter. To maintain a constant perfusion rate and keep the filter in the bioreactor free of debris, 24 hours after the seeding we placed an inline filter between pump B and the reservoir, and reversed the direction of the cross-flow while maintaining the same value of the perfusion or crossflow rate. We kept this direction of the cross-flow for the duration of the experiment. For five-micron pore-size DURAPORE filter (Millipore Corp., Bedford, MA) in the bioreactor, we used a 0.8/0.2-pm pore-size double layer inline syringe filter (Pall Corporation, Ann Arbor, Ml). In this way, cell debris capable of clogging the filter in the reactor was captured in the inline filter. By replacing the inline filter every 72 hours, we managed to maintain a constant "bottom up" cross-flow through the channels with cells. This helps clean the filter out of debris and maintains the crossflow or perfusion through the tissue. Figure 4-4 shows the removal of debris from the bioreactors as a result of Techno1o' Massachusetts MassachusettsInsdtute Institute of ofTechnology 99 - 99 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center the reversal of crossflow, 24 hours after cell-seeding into the channels of the reactor. In the absence of such a flow reversal, the crossflow through the tissue (the pull-down crossflow) is seen to fall to zero quickly past 24 hours, possibly due to the clogging of the Durapore membrane filter placed between the two silicon scaffolds. Cleaning Effect of Crossflow (a) (b) - 45 min. post flow reversal Figure 4-4: Cleaning effect of crossflow Removal of cell debris from the membrane filter sandwiched between the silicon scaffolds, as a result of reversal of crossflow 24 hrs. after cell seeding (a) before reversal and (b) after reversal of crossflow 4.4 Evaluation of hepatic phenotype: albumin and urea secretion In order to assess the basic hepatic phenotype of the tissues formed in the MilliF reactor, albumin (Figure 4-5) and urea secretion rates (Figure 4-6) were measured in the bioreactor cultures. Bioreactors were assembled and seeded with Day 3 spheroids and cultured upto Day 14 (post cell isolation). Massachusetts Massachusetts Institute Institute of of Technology Technology -100- 100- Biotechnology Process Engineering Center Biotechnology Process Engineefing Center ALBUMIN 35 2 30 H_= R2 -0.997 -_ _ ·I:· .. 25 20 15 ;91· bi 7.82 t 0.51 n: 3 10 5 0 0 nged 2 4 6 8 r.4 I_, :, 'A - Z 10 , ~ 12 14 "I,' Figure 4-5: Albumin secretion rates in bioreactor cultures Serum albumin concentrations of medium samples were determined by a sandwich enzyme-linked immunosorbent assay (ELISA) [105]. Polvclonal sheep and-rat albumin IgG (ICN, Costa Mecsa, CA) and horseradish peroxidase-conjugated goat anti-rat albumin lgG (Accurate Chemical, Westbury, NiY) were used in these assays. Absorbance values were measured with a SpectraNAX 250 microplate specrrophotometer (Molecular Devices). Total albumin content in bioreactors or static cultures was determined by multiplying the measured albumin concentration by the total medium volume at the time of sampling. Urea concentration was assessed bv Berthelot determination methods (urea nitrogen kit from Sigma, procedure 640). Both the urea as well as the albumin data was normalized using measured values of total DNA from the reactors (See Appendix for protocol). of '!'echnolngv Nlassachusetts Institurt: Mmassachusetts Instiuurc of'lechnology - 101 101- Biotechnology Process Engineering Center Biote~chnologyProcess Enineering Centerr , 40'--~- < -1 u o 120 -: i o :3. .100 C i I '0o 80 60 40 -) t o40 - E 0 O------0 2 ; 4 6 8 10 = 12 14 Days in Culture Figure 4-6: Urea secretion rates in 3D bioreactor cultures Both albumin as well as urea secretion rates measured in the MilliF bioreactor were comparable to those seen in the macroreactors (Figures 4-5 and 4-5). In addition, on the basis of urea and albumin secretion rates, it may be cautiously concluded that the tissue phenotype in the MilliIiFreactors are comparable with those seen in the macroreactors. 4.5 Ultrastructural evaluation of tissue phenotype Transmission Electron Micrographs of the tissue sections were prepared by standard protocol. The electron microscopy was done in collaboration with Dr.Donna Stolz of the University of Pittsburgh Medical Center. Scaffolds were fxed in 2.5/o glutaraldehyde in PBS for 24 hr., washed three times with PBS, and post-fixesd for 1 hr with aqueous 1% osmium tetroxide. After three PBS washes, scaffolds were dehydrated through a graded ethanol series, and then further dehydrated in four 15-min changes of 100% ethanol. Scaffolds were subjected to two 10-min incubations in propylene oxide, and then preembedded with a 1:1 mix of propylene oxide: Polybed 812 epoxy resin (Polysciences, Warrington, PA) for 1 h. Scaffolds were then incubated in 100% Polybed overnight at 40 (,. The following day, resin was changed technology Massachuserts Institute Massachusetts nstitute of of'Technology · ___II__I__I__I___C·___ 102-- 102 - Biotechnology Process Engineering Center iotechnolog. Process Engineering Center -- four times before embedding chips in a thin layer of resin, just enough to fill the channels, and curing at 370 C overnight and then at 650 C for an additional 2 days. Cells growing within the channels were removed from embedded chips by rapid, alternating treatments in liquid nitrogen and boiling water. This treatment shattered the scaffolding and allows for embedded cells and channels to be removed intact. Blocks of cells were re-embedded in rubber molds and cross-sectioned perpendicular to channel flow. Thin sections (60 nm) were collected on copper grids and stained with 4% uranyl acetate in 50% methanol for 7 min and with 1%'olead citrate for 10 min. Cells were viewed with a JEOL (Tokyo, Japan) JELI 1210 transmission electron microscope (TE'M at 80 kV. Figure 4-7 shows a representative TENMof bioreactor tissue thin sections. 1 micron I micron '(IhalltCEge BC 5 Cell -Cell microns J Ulctil. ER. . ER .: I ECM _0 I...., N ' N , 1r .:tk.$, _;L-- ~s-. _ Figure 4-7: TEMI's of tissue phenotype in the bioreactor. Presence of a large number of mitochondria (NI), rounded nuclei (N), bile canaliculi (BC), endoplasmic reticulum (ER), extra cellular matrix material (ECM) and cell-cell junctions are indicative of a healthy tissue phenotype. Scanning Electron Mlicrographs of the tissue structures formed in the reactors are presented in Figure 4-8. The presence of a large number of microvilli (M in Figure 4-8, (e)), are indicative of the presence of metabolically active, functional hepatocytes. The presence of some endothelial cells at the interface between the hepatocytes and the fluid flow conduit in the center of the channel (Figure 4-8, (a), (b)) is suggestive of heterotypical re-organization. In some cases, a layered-rearrangement of different cell-types is seen in the tissue, reminiscent of ~\ lassahusetts NIasachusett-s Institue o~fTechnologv n~tirurcot - 13 Technlogy - 10)3- ngineerig Cente BiotchnologyProcess 3iotechnolog Pocess Engincering Center the in ivo tissue phenotype (See Figure 4-8, (c), (d), (e)). However, these results are not typically seen in all cultures and detailed quantitative histological and microscopic studies will be needed to characterize the localization of the various cell types present in the hepatic tissue formed in the 3D bioreactors, once a technique to identify and characterize the various cells that constitute the tissue phenotype is developed. Figure 4-8: SEM's of tissue structures formed in the micro channels of the bioreactor Bioreactor cultures were prepared at Cambridge, KN[and the scaffolds with the tissue structures were fixed and shipped to Pittsburgh. For the SEIM's on the bioreactor, scaffolds were removed from the bioreactor and fixed in 2.5%ioglutaraldehyde solution for I hour, and washed three times with PBS, each wash lasting for 15 min. The scaffold was then incubated with 1 Osmium Tetroxide for 1 hour, followed by 3X PBS washes (15 min each). The scaffold was then fixed in Thiocarbohydrazide (TCH) for 30 min, followed by three successive washes (15min each) in PBS. The scaffold is fixed in Osmium tetroxide for hour. This is followed by another set of PBS washes (15 min, 3X). The scaffold is once again fixed in TCH for 30 min, followed b the PBS washes as before. This is followed by a final Osmiun Tetroxide fixing step (1 hour) followed by the 3X PBS washes as before. Serial ethanol Biotechnology Process Engineering Center Biorcchnology Process Engneering Center Technology Institute of i'vL'issachusctts Ms~sachusettsInstitute ofTec~nology - -~~~_ ··I dehydration steps (30°'o, 500%/,70%, 95°'o Ethanol for 15 min each) followed by four successive incubation steps of the scaffold in Hexamethylenedisilazane (HMDS)-ethanol solution (250o, 50%, 75% and 100% HNMDSsolution in ethanol, 15 min each). Most of the HMNDSis aspirated out of the dish till a very small laver of solution barely covering the scaffold is left behind. The scaffold is stored in a desiccator till completely dry. Histological sections stained with toluidine blue are shown in Figure 4-9. Viable cells take up the toluidine blue stain and are colored dark blue. Serial sections taken from many different channels of bioreactors from different experiments confirm the presence of mostly healthy and viable cells in the tissue. OUS Ithy ue: jidine e staining j. 50 -mi t. ,~~~~~~~~~~I Figure 4-9: Toluidine blue stained sections of the liver tissue from a bioreactor channel 4.6 Maintenance of p450 isoforms in the 3D bioreactor culture As described in Chapter 1, the maintenance of the basal expression levels of the CYP450 gene and protein expression is an important pre-requisite of any in vitroscreen used for predictive drug metabolism and toxicology. The maintenance of the basal gene and protein expression of a number of CYP450's of Pharmaceutical relevance was assessed in the MilliF 1echnologv NIasachusetts Mlassachuserts lnsritute Instiute of of Technology 105 - 105- Biotechnology Process l5nginccring C±nter Botechnology Process Engineerng Center bioreactor. While the eventual goal is to be able to measure and quantify the actual rate of metabolism of drug-like compounds in the bioreactor, preliminary experiments focused on assessing the ability of the bioreactor to maintain the expression of the Phase I drugmetabolizing enzymes (CYP450's) in the bioreactor culture (Figure 4-10), and the ability of the culture to induce CYP450's in response to the addition of prototypical inducers by Western Immunoblotting (see Appendix for detailed protocol). In addition the repeatability of the cultures system was tested across different experiments as well as across bioreactor cultures I 'I-l~~ from the same experiment. Cells after Reactors isolation CYP2B1, ' CYP2B Day3 Day7 Spheroids 10 13 Figure 4-10: Maintenance of expression of CYP450 2B protein for at least upto two weeks in bioreactor culture. (All lanes have beennormalied for the same amount of totalprotein) The expression of both Phase I and Phase 11enzymes were seen to be very repeatable across different bioreactor cultures (bioreactors from both the same experiment as well as from multiple experiments showed comparable expression of CYP proteins as well as Phase II proteins). Figure 4-11 shows the repeatability both across experiments as well as across different bioreactor cultures, of the expression of an important Phase I protein, CYP4503A2 and a Phase II enzyme UGT1 A (UDP glucuronosyltransferase-lA). of Technology Institute of Massachusetts Institute Mlassachusetts Tchnology ·-- - 106- - 106- ------------------ Biotechnology Process Engineering Center --Biotechnology Process Engineering Center Expt.1, Expt. 2 Rxtr 1, Rxtr 2 n ," :7 Day 7 M'm 7 - av - Y Dhoeo rI IILO 11 I11 enzyme: UGT 1A CYP3A2 Figure 4-11: Repeatability of p45 0 expression in bioreactor cultures The bioreactor cultures are seen to be pretty repeatable in their expression of kev Phase I and Phase II ezvme proteins (Westerns), both across reactor cultures as well as across experiments. 4.7 Qualitative spectroscopic measurement of p450-lA activity A specutrometric system, designed in hourseby Dr. Karel Domansk 3 , (See Figutre 4-12), was used to measure in situ, the maintenance of cytochrome P450 1A (CYP4501A) in bioreactor-cultured rat hepatocytes. thermalshield lightsourcerEX filter metallcwavegude aluminumheal sink ng bases&slandoffs Figure: 4-12: Spectrometric system used for in .rsitu spectroscopyr of the 3D perfused tissue (Photographly Dr:Karte/Domansk-y) nsiut o ichokg: Masahuern nstitute of Technolog Mlassachusetts 1)7- - I107- ioehnloy roes ngnerig ene Biotechnology Process Engineering Center EROD assay for P4501A i o EROD A Assay n ' -1 Phase II Conjugates Resorufin Ethoxyresorufin * 120 min. exposure to 8.3 FM 7-ethoxyresorufin, 10 p[Mdicumarol * 48 hr. induction, 5 tM 3-methylcholanthrene Figure: 4-13: The EROD Assay to measure the activity of P4501A Ethoxvresorufm has low fluorescence but it is transformed into a highly fluorescent form, resorufin, in the presence of CYP4501A. This is the standard Ethoxyresorufin-ODealkylation (EROD) assay shown in Figure 4-13. The absorption and emission peaks of resorufin as well as the optical filters we used in the assay are shown in Figure 4-14. First, the signal from a primed bioreactor with only the albumin-free cell culture media and without the cells, was measured (Figure 4-14 b, black line). This is essentially the bleed through of the optical filters. Then, the signal from a bioreactor culture with the tissue structure but without any Ethoxy Resorufin added was measured (blue line). The small increase in the intensity level can be attributed, for example, to autofluorescence of the cells. Then, ethoxyresorufminwas added to the cell culture medium at a high substrate concentration (10 IM)and the fluorescent product resorufin (red line) one hour after adding the substrate. In the control experiment (Fig. 4-14c), a bioreactor without cells was used and ethoxyresorufin was added to it prior to the measurement of signal. nttt fTcnlg Mascuct Massachustts Institute of Technology 08BoehooyPoesEgneigCne -108-- _____ Biotechnology Process ngineering Center __··___ b) Intensity (counts) Spheroids Day 3 500 40( Hepatocyte growth 300 medium a) _______a) ethoxyresorifin 5 I1A IA··- 200--(no cells) 200 resorftrill 100 I ii 500 I ell, , I 550 600 Wavelength C) Intensity (counts) * ~~~C) 00 400 650 (nm) 700 Control measurement without cells I II 300 Hepatocyte growth 200 medium 100 (no cells) i i 0 I 500 550 600 650 700 wavelengtn nim) WAVELENGHT[nm] Figure 4-14: Detection of CYP activity using a fluorescence detector (a) The emission and excitation spectra of Resorufin, with peaks at 571 nm and 585 nm respectively. (b) Signal due to the formation of resorufin in the bioreactor as a result of the addition of the C(\T1YAsubstrate -Ethoxvresorufin. (c) Control reactor with cells but without the addition of Ethonvresorufin.(Experiment dlonewith ResearchScientist Karel Dovnansky) 4.8 Summary of key results and conclusions Bioreactors were cultured with primary rat hepatocytes upto two weeks in culture. Albumin and urea secretion rates were seen to be comparable with rates previously reported in the macroreactors. Preliminary microscopy data and electron micrographs reveal the presence of a viable, healthy hepatic tissue phenotype in the channels of the bioreactor. The tissue constructs formed in the bioreactor are seen to maintain the basal expression of many of the important, pharmaceutically relevant CYP450's upto two weeks in culture. The bioreactor fluidics was also optimized to maintain a constant perfusion crossflow rate through the channels of the bioreactor. The activitv of one of the (C-1Y450'swas qualitatively measured using a spectrophotometer and a non-fluorescent substrate Ethoxy Resorufin that is converted to a fluorescent product Resorufin. Technology of TechnologyMassachusetts Institute Maiachuserts Institlre of - 109 1(9 - - Biotechnology Process Engineering Centcr Biotechnology Process RnngincringfCent=r Chapter 5 Measurement of Xenobiotic Metabolism in 2D and 3D Cultures 5.1 Challenges in the measurement of intrinsic reaction rates in a 3D system The basic design of the microfabricated bioreactor scaffold-sandwich that houses the tissue constructs was explained in detail in Chapters 2 and 3. While the three-dimensional nature of the tissue helps recapitulate aspects of the in zito tissue morphology, it may also lead to the creation of concentration gradients within the tissue of specific metabolites, media components, products of metabolism, and xenobiotics, due to the presence of a duffusional mass transfer resistance in the 3D tissue. Thus the experimentally measured apparent rates of metabolism of xenobiotics will be different from die intrinsicrates of metabolism within a single cell. The mass transfer model described in detail in Chapter 3 may be used to predict the tissue concentration distribution of metabolites, using appropriate parameter values for the Thiele modules (or the Damkohler Number) and the Peclet number. The same mass transfer model may also be used to predict the itrinsic rates of metabolism, given the apparent rates of metabolism calculated experimentally. For the purposes of this thesis, all experiments were performed using high substrate concentrations of drug compounds (xenobiotics). Thus the theoretical value of the Damkohler numbers (thiele modulus) were sufficiently low, so that the enzymatic reaction rate in a cell in the tissue was the rate determining step at steady state (i.e. the after characteristic time scale for diffusion of the drug to the enzyme site was reached, typically on the order of a few minutes for a tissue length scale as large as 200 tm). This is explained in detail with specific model predictions in Section 5.6. In addition, while measuring the specific hydroxylation rates of hydrophobic drugs, a significant problem is posed by the drug binding to albumin in the media and to various parts of the reactor and fluidic circuit. Thus, it is imperative that albumin-free media be used for all Technology of 'echnology Institute of Massachusetts Institute Mlassachusetts -- '- 110- 110- Biotechnology Process Engineering Center Biotcchnology Process Engineering Center -----' drug-metabolism measurement studies. In addition, paralyne coating of the reactor fluidic circuit helps minimize the adsorption of testosterone to the reactor and fluidics. Also, it is imperative that the total volume of the reactor and fluidics be minimized to ensure that the system is well-mixed during the course of the drug metabolism studies, when multiple samples at different time points may be needed to validate the linearity of the Michaelis-Menten enzyme kinetics. A well-mixed system allows for small sample volumes to be taken at multiple time points from any port/ location of the reactor and its fluidics. A wellmixed system with a small average residence time of a metabolite, allows for systemic equilibration of local concentration changes over a time scale much smaller than the time between two consecutive sample points. On the other hand, once the linearity of the kinetics has been established, a single sample maybe taken at the end of the experiment for any new experiments. If the entire reactor fluidic volume is collected, there may not be a need to ensure that the reactor is indeed well-mixed - as the collection of the entire reactor volume would ensure that all of the products of metabolism are completely collected and quantified cumulatively at a single time point, so long as the Michaelis-Menten rate of product formation is linear upto that time point. Minimizing the reactor and fluidics dead volume also helps reduces the loss of products of metabolism to the unusable non-organic portion of the media through downstream vacuum drying and other sample processing techniques prior to its injection into an HPLC-UV separation and detection system. The quantity of the un-usable non-organic portion of the media solution has been known to scale with the quantity of media. Thus, minimizing the volume of the fluidic circuit can help increase sensitivity of the assay by minimizing loss of products of metabolism to the non-organic phase during solvent extraction. 5.2 Paralyne coating of the fluidic circuit A large fraction of the hydrophobic drugs such as testosterone and diazepam that were used as substrates for the CYP450's in the bioreactor, adsorbed to Silicone® and CFLEX® tubings used in the reactor fluidic circuit. In addition, the drugs adsorbed to the inline filter and a negligibly small fraction adsorbed to the Durapore® filter used in the scaffold sandwich. Technology Massachusetts Miassachusetts Institute nstitue of ofTechnology -III -111- Biotechnology Process Engineering Center Biotechnology Process Engineering Center - More than 70% of the hydrophobic drug, was lost through a combination of non-specific binding and adsorption in empty control reactors with no cells seeded in them (Figure 5-1). A number of different tubing materials were evaluated - however, the tubings that were seen to be inert to the hydrophobic drugs and thus did not bind/ adsorb/ absorb them, were inflexible and hard - properties that made them unsuitable for use in peristaltic pumps. The fluidic circuit in the bioreactor set up consists of CFLEX® peristaltic pump tubing and Silicone® tubing. The Silicone tubing was replaced by an inert Teflon® HPLC tubing, to eliminate loss of drug to these tubings. The peristaltic CFLEX® pump tubing was passivated by a layer of paralyne that was coated by a vapor deposition process at Advanced Coating, Inc. (Cucamonga, CA). Drug/ Substrate adsorption to silicone and CFLEX tubing Adsorption of diazepam and its metabolites to silicone tubing a SII II 0 iiL C C 0 0Ca,U 0 20 40 60 80 100 120 140 160 180 200 Time after addition of drug Figure 5-1: Loss of hydrophobic drug to adsorption, absorption and non-specific binding in empty reactors with no cells seeded Parylene is a common generic name for a unique series of polymers based on paraxylene. The three most common types of parylene are referred to as: Parylene N, Parylene C, and Parylene D. The parylenes are formed by the pyrolysis of a di-p-xylene (dimer) in a Technology Institute Massachusetts ~assachusetts Institute of of Technology -112- -112- Biotechnology Process Engineering Center Biotechnology Process Engieering Center vacuum environment which is then deposited on a cooler (i.e. room temperature) substrate under continuous vacuum. Vapor phase deposition of the parylene polymer allows it to be formed as a structurally continuous film which is truly conformal to the design and structure of the substrate upon which it is being deposited. Paralyne N, with a chemical structure as described in Figure 5-2, was identified as a suitable passivating agent as it affords the greatest surface protection. Figure 5-2: Parylene N was used to passivate the pump tubings. The parylene N polymers is deposited from the vapor phase by a process which in some respects resembles vacuum metalizing. Unlike vacuum metalization, however, which is conducted at pressures of 10-5 torr or below, the parylenes are formed at around 0.1 torr. Under these conditions the mean free path of the gas molecules in the deposition chamber is in the order of 0.1 cm. Therefore, unlike vacuum metalizing, the deposition is not line-of-sight and all sides of an object to be encapsulated are uniformly impinged by the gaseous monomer. This is responsible for the truly conformal nature of the coating. The passivation process consists of three main phases - the vaporization, pyrolysis and deposition processes as described in Figure 5-3. - 113MassachusettsIns~~~~~tuteof Techno~~~~o~~t - 113Massachusetts Institute of Technology Biotechnology Process Engineering Center~~~~~~~~~ Biotechnology Process Engineering Center OAIFour _Or~A4nM W VkLq4-%Mvxfi IDIOMq YVAPMOE VACUU Pim Figure 5-3: Steps in the conformal deposition of Parylene N on CFLEX ® tubing Parylene thickness is a function of the amount of vaporized dimer and chamber dwell time and can be controlled accurately to within +/- 5% of targeted thickness for most typical applications. A coating thickness of at least 100 im was used during the deposition process. Due to the nature of the deposition process, the thickness of paralyne was seen to taper towards the center of the tubing. Thus, short tubing lengths of not more than 20 mm were used to ensure a uniform pin-hole free coating. In addition pump tubings less than 0.03" could not be passivated uniformly - hence a crossflow pump tubing of 0.03" ID was used. Massachusett nsiteoTch lgy- Massachusetts Institute of Technology 4- -114- Bitcnlg Prcs nieeigCne - - Biotechnology Process Engineering Center Paralyne coatin of tubino to minimize/ prevent adsorption Adsorption of testosterone on paralyne coated tubing 1.2 0 byI | . · - -- - -- E i ---- II C 0.8 IC 0.6 0.4 O C 0 0 0.2 CO) 0 0 100 200 300 400 500 600 Time (min) Figure 5-4: Minimal loss of hydrophobic drug to Parylene N coated tubing An empty reactor without cells now assembled with the Parylene coated pump-tubings and the Teflon" tubings were seen to absorb/ adsorb/ bind to not more than 2% of the hydrophobic drug as well as its products of metabolism (Figures 5-4 and 5-5). Massachusetts Massachusetts Institute Institutet of of Technology echnology 115-115- Biotechnology Process Engineering Center Biotechnology Pocess Engineering Center Paralyne coating of tubing to minimize/ prevent adsorption Adsorption of products of testosterone metabolism on paralyne coated tubing 1.2 ;i C _ __-----------___ 'U 2 0.8 Cr C 0.6 O 'IC) 'U 0.4 0.2 0 0 100 300 200 400 500 600 Time (mln) Figure 5-5: Minimal loss of hydrophilic products of metabolism due to Parylene N coated tubing 5.3 Minimizing the dead volume in the bioreactorand fluidics Three concurrent strategies were employed to minimize the dead volume in the bioreactor. The advantages and disadvantages of each of these strategies are discussed in detail below: 5.3.1. Reservoirless fluidic circuit The original design of the bioreactor fluidic circuit included a reservoir with a dead volume of 15 ml and a tubing network with a dead volume of nearly 2 ml. A re-designed fluidic circuit that did away with the reservoir, so that the dead volume in the tubing acted as the "reservoir" instead, was used in an attempt to minimize the total dead volume of the fluidic system. Figure 5-6 shows the re-designed fluidic circuit. Additionally, smaller bore Technology Massachusetts TNlasachusetts Institute Institute of ofTechnology -116-116- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Teflon® (0.007", Alltech, PA) tubings were used in a very large portion of the tubing network instead of the 0.062" silicone tubing. Minimization of dead volume in Reactor and Tubing Original Configuration - Reactor and Fluidics Reactor + Tubing dead volume: 17.5 ml Reservoir less Configuration - Reactor and Fluidics Reactor + Tubing dead volume: 360 ul Figure 5-6: Reservoir-less reactor fluidics configuration to minimize dead volume 5.3.2. Small reservoir, small bore tubing fluidic circuit While the reservoir-less reactor configuration helped minimize the total reactor and fluidics dead volume, it posed serious oxygen equilibration and de-gassing issues. Exchange of oxygen between the ambient air and the media in the tubing, was minimal as the Paralyne coating minimized the transport of oxygen through the tubing. Additionally Teflon® also has low oxygen permeability. Thus, in the absence of the reservoir, there was a possibility that the media solution used in the reactor fluidics was not saturated with oxygen, as it entered the bioreactor ports. In the earlier fluidics design, oxygen exchange was made possibly by the presence of both oxygen permeable Silastic®tubing as well as a 15 mL reservoir. Massachusetts nstitute ofTechnology -117- Biotechnology Process Engineering Center To address this issue, the fluidic circuit was re-designed to now incorporate a conical glass (oxygen permeable and with a re-sealable Teflon septum) reservoir that housed 2 mL of media saturated with oxygen following equilibration with the ambient air, while continuing to use the low bore size Teflon ® tubing. The use of very small diameter tubing in the reactor fluidics necessitated the use of medium pressure rated HPLC fittings (Alltech, PA) Figure 5-7: Components of the two pump, small reservoir fluidic circuit (a) The Paralyne coated pump tubing and small bore Teflon® tubing circuit (b) A view of the microreactor in the fluidic circuit (c) The medium pressure water union HPLC fitting and inline debris collection 0.8 [tm/0.2 itm filter and (d) the small-volume conical glass reservoir with a re-sealable Teflon® septum on top. Inttt fTcnlg Mascuet Massachusetts Institute ofTechnology -11-118- Prcs nieeigCne B=tcnlg Biotechnology Process Engineering Center The reduction in the total dead volume of the reactor and fluidics helped improve the sensitivity of detection of the system, especially for metabolites that were produced in very small amounts, in response to the addition of specific drugs to the system. 5.4 Measurement of xenobiotic metabolism in 2D collagen sandwich (2DCSW) and 3D Bioreactor (3DB) cultures Saturation concentration of a typical xenobiotic (i.e. a concentration at which the Michaelis-Menten rate is zeroeth order with respect to substrate concentration) were used in the measurement of specific p450 enzyme activity in the 2D collagen sandwich cultures as well as in the 3D bioreactors. Testosterone was chosen as the candidate xenobiotic, as it is metabolized into many different hydroxylated products by a large number of CYP450's [106]. Quantitative analysis of metabolism in in-vitro cultures Measurement of Enzyme activity using candidate drugs/ substrates OH Metabolism of Testosterone 1l-OH-testosterone H i\-- .8I %.e 2a/p-OH-testosterone l' - 6P-OH-testosterone 1%-~ _ . . OH 4,1/.Z 1 6-0O H-testosterone 15D-O H-testosterone Not a major metabolite in isher rats (Caldwell et al, androstenedione 1996) Measurement of Steroid hydroxylation, Methods in Enzymology, VoL206,Sonderfan, Waxman et al : Sprague-Dawley Figure 5-8: Testosterone biotransformation pathway in the rat liver Technology Insfitute of Massachusetts Institute Massachusetts ofTechnology - 119- -119- Biotechnology Process Engineering Center Biotechnology Process Engineering Center In addition, testosterone metabolism has been well characterized and understood in literature in a number of different in vitrocultures [86, 106-120]. The important components of the biotransformation pathway of testosterone are reproduced in Figure 5-8. Two dimensional collagen sandwich cultures were prepared similar to previous methods suggested by Dunn and co-workers and later reviewed extensively in literature [4, 8, 18, 24, 44, 121, 122]. 35 mm tissue culture treated six-well plates (Falcon, USA), were coated with 600 1l of a collagen mixture (4.7 ml sterile water, 1.3 ml of modified PBS solution [lg glucose, 1.8 5g sodium bicarbonate, in 50 mL PBS solution], 6.9 ml of a 3mg/ml collagen (type 1) solution (Vitrogen, USA)), and with pH of solution adjusted to - 7.4. Plates were coated overnight to allow the collagen to gel in an 8.5% C02, 37'C environment. Percolled primary rat hepatocytes were seeded at a density of 50,000 cells/ cm2 , and evenly distributed on the plate, in lmL of Hepatocyte Growth Medium (H-GM). Following an overnight incubation at 37°C and 8.5%C02, that allowed the cells to attach to the substratum, the media was aspirated from the plate and 300 ul of the collagen mixture was added to form the second layer of the sandwich. The collagen was allowed to gel over a 45 -60 min period, following which 1 mL of HGM was added to the well. The cultures were incubated in a controlled 37°C, 8.5%CO2 environment. Media were changed everyday. 3D Bioreactors were assembled and seeded as described in Chapter 4, Section 4.3. On the day when xenobiotic metabolism experiments are to be performed on the 2D culture and the 3D bioreactor, fresh albumin-free Hepatocyte Growth Media (HGM) was added to both the 2DCSW as well as the 3DB cultures. The 2DCSW cultures were shaken gently on a shaker kept inside an incubator that provided a controlled 37°C, 8.5%C02 environment. Media was changed twice in the 2DCSW at 30 minute intervals to help wash out the albumin present in the collagen. This can help minimize the loss of drug or xenobiotic due to equilibrium binding with albumin. Media were changed twice, again at 30 min intervals, in the 3DB to dean out the rat albumin present in the system. 250 VM concentration of testosterone, that corresponds to a concentration that saturates the p450's involved in the metabolism of testosterone [117, 118, 120] was made up in fresh albumin-free media solution, and was added to the 2DCSW Massachusetts Massachusetts Institute Institute of of Technology Technology -120- -120- Biotechnology Process Engineering Center Biotechnology Process Engineering Center cultures as well as the 3DB. Products of metabolism of testosterone were collected 60 min. after the addition of the substrate to both the 3DB and the 2DCSW cultures. The use of the shaker with the 2DCSW helped eliminate concentration gradients in the media. Linearity of the Michaelis-Menten kinetics was verified over this time-span. To the samples from the 2DCSW and 3DB cultures, a known concentration of an internal standard (Methyl Testosterone) was added. The samples were them vacuum dried (SpeedVac, Thermo Electron Corporation, hA) and testosterone and the hydroxy-products of testosterone were extracted from the dry powder using methanol as solvent. A schematic of the sample processing steps is detailed in Figure 5-9. HPLC-UV detection and quantitation of hydroxylated products Quantifying intrinsic rates of metabolismof testosterone in 3 Bioreactors - Recycle Mode, and in 20 sandwich cultures AM >> Km c/reeunadsorbed = 250 testosteronem .. .. 11 HPLC- UV Detection Samplef of reactor or ZD cultures internal std. Concentrate scample under vacuum - dry 1to a powder meTQDoIIsm Una ilTernal std using 50 -100 ul methanol 2 5 6IP-OH Internal Androstenedione standard ** a chosen: Methyl 16a-OH Measure concentration of products as -Testosterone a function of time. 16P-OH id min. 35 min. 40 min. Time point: 60 min after addition of substrate, verified to be within the linearity of M-M kinetics 46 min. Figure 5-9: Steps in quantifying the concentration of hydroxylated products of testosterone metabolism using HPLC-UV. Technology Massachusetts Massachusetts Institute nstitute of ofTechnology - 121 121- - Biotechnology Process Engineering Center Biotechnology Process Engineeding Center Quantification of the concentrations of testosterone and the products of metabolism of testosterone was done by HPLC analysis on an Agilent 1100 series (Agilent Technologies, Waldbronn, Germany) which consisted of a 9725 Rheodyne injector (Rheodyne, Rohnert Park, CA) and a G1314A variable wavelength UV detector at 240 nm (Agilent Technolgies). An aliquiot of the extracted sample (5uL) from each incubation was injected on a Capcell Pak C18 column type UG120 size 2.0mmID X 150mm, 5um particle size; Shiseido Fine Chemicals, Tokyo, Japan and eluted at a flow rate of 200ul/minute by the gradient with the mobile phase, which consisted of solvent A (20 mM ammonium acetate in 10% methanol) and solvent B (90% methanol). The solvent gradient (solvent B) used for eluting testosterone and its metabolites was as follows: 0 min, 10%; 0-10min, 10%10-20min, 30%; 20-30min, 55%; 30-38 min, 55%; 38-45 min, 100%; 45-50 100%. The gradient was then returned to the initial condition (10% B) and held for 8 minutes before analysis of the next sample. A standard curve of the ratio of AUC units of testosterone (or products of its metabolism) to that of the internal standard, was plotted against known concentrations of the standards. Standards were all purchased from Steraloids, RI. Table 5-1 lists the slopes of the standard curves generated using known concentrations of the hydroxylated testosterone standards as well as the testosterone standard. A specimen standard curve is reproduced in Figure 5-10. The slopes in Table 5-1 were generated using standard curves such as that reproduced in Figure 5-10. A known concentration of internal standard - 32 M of Methyl testosterone - was added to all the standards to normalize the data and to remove any technical errors, associated with the sample preparation steps. Methyl testosterone was chosen as the internal standard as its physical properties are very similar to those of testosterone and its hydroxylated products. At the same time, Methyl testosterone is stable and inert and does not normally react with testosterone or the hydroxylated products. Technology Massachuse Mlassachusetts Institute Institute of ofTechnology -122-122- Biotechnology Process Engineering Center Biotechnology Process Engineerig Center 6-alpha OH testosterone 2A1 32.208 0.706 15-alpha OH testosterone 2A1, 2C12, 2C13 33.850 0.742 6-beta OH testosterone IAl, 2A1, 2B2, 2C11, 2C13, 3A1, 3A2 34.535 16.141 0.757 7-alpha OH testosterone 2A1 35.721 16-alpha OH testosterone 2B1, 2Cll, 2B2, 2C7, 2C13 35.904 11-alpha OH testosterone -N/A- 36.634 16-beta OH testosterone 2B1, 2B2, 3A1, 3A2 36.725 2-alpha OH testosterone 3A2 38.002 0.833 2-beta OH testosterone 3A1, 3A2, 1A1, 1A2 38.550 0.845 15-beta OH testosterone 3A1, 2A1 Androstenedione 2A1, 2B1, 2B2, 2C11, 3A1, 3A2 0.783 26.848 0.787 0.803 28.917 0.805 40.283 8.169 0.883 Testosterone 42.975 42.000 0.942 Methyl Testosterone (IS) 45.621 1.000 Table 5-1: Products of testosterone hydroxylation and p450's that mediate their formation. * Relative retention time is defined as the ratio of Retention time of a given metabolite dividied by the retention time of the Internal Standard # from [106, 120, 123], shaded cells indicate hydroxylation products whose rates were quantified as part of this thesis. Table 5--1also lists the approximate retention times of the hydroxylated products as well as the relative retention times of the various metabolites with respect to the internal standard. The absorbance spectrum of each individual metabolite was compared against the spectra of the standards to validate the detection of any hydroxylated product. A sample Mascuet nttt fTcnlg Massachusetts nstitute of Technology 1 3-BoehooyPoesE -123- gneigC ne iotechnolgy Process Egineering Cente I spectrum of one of the hydroxylated products - 6P hydroxy-testosterone is shown in Figure 511. 6-beta hydroxytestosterone t = 34.535 min. 4.0 I y = 0.0162x R2 = 0.9993 h 2.0 U 4 1.5 i o 0.5 Fit 0.0 0 20 40 I 60 80 100 I 120 ] 140 Concentration (uM) Figure 5-10: Specimen standard curve used to calculate the normalized slope of AUC ratio (UV peak absorbance of metabolite divided by the UV peak absorbance of internal standard of known concentration) versus concentration of metabolite. The slope thus determined was used to calculate the concentration of hydroxylated product in the sample. The calculated concentrations of the hydroxylated product are then converted into an average rate of product formation using experimentally determined values of the total cell number per unit volume in the culture (usingmeasuredvaluesof totalRNA or 18sgenein cltatreand a cellnumberversustotalRNAI or 18sgenestandardcurve- seeAppendix and Section6.2), and the total time over which the drug or xenobiotic was administered to the culture. itcnlg rcs niergCne Inttt ofTcnlg ~ ~ ~ ~ ~- 14 Massachusetts~~~ 124Biotechnolgy ProcssEngineering Center Massachusetts Institute of Technology Peak :8 at 34.535 min Name : 68hudroxutestosterone : 200 225 250 275 300 325 350 375 n Figure 5-11: Specimen absorbance spectrum of a hydroxylated product and recorded retention time. The spectrum and the retention time are unique for each metabolite detected. 5.5 Theoretical validation of the zeroeth order Michaelis-Menten Kinetics at high substrate concentrations As described in Chapter 3, the metabolite concentration gradient set up in the tissue is a function of two main parameters - the Peclet Number that characterizes the crossflow through the channels and the Thiele Modulus or the Damkohler number for the metabolite. The Peclet number in the channel is sufficiently high (Pe > 100) so that convective transport dominates, and the boundary layer set up at the tissue fluid interface is of negligibly small thickness. For the case of testosterone, the largest value of K, among all enzymes that are involved in the biotransformation pathways, is on the order of 50 [M. Thus, the concentration of testosterone used in the experiment, namely 250 }xM,is atleast 5 times the value of the largest K, It is therefore expected that the kinetics of product formation should be zeroeth - 125Institute of Technology Massachusetts -125Massachusetts Institute of Tchnology Biotechnology Process Engineering Center~~~~~~~~~~~~~~~~~~~~~~~ Biotechnology Process Engineering Center order with respect to substrate concentration. Additionally, the large value of the substrate concentration results in a maximum value of thiele modulus of I)2 = V x DC0 n, = 0.03 for the enzyme with a K, of 50 M (assuming a tissue length scale (L) of 100 Jim (Chapter 3), substrate diffusivity (D) of 2 x 10-5 cm 2 /sec [96], a volumetric tissue cell density (n,.) of 4.0 x 1013 cells/m3 (Chapter 3, Figure 3-9) and a maximal enzyme activity (,,) reported in literature of 1300 pmoles/106 cells/min [50]). Since the value is significantly less than 1.0, the mass transfer resistance to the transport of testosterone to the individual cells in the tissue is negligible, so that there is hardly any concentration gradient set up in the tissue. The schematic in Figure 5-12 describes such a scenario. brug Metabolism in the Bioreactor - the limiting case Q- . Single Channel Side view Q,..c- 1 vrVDAn "I ntracellular" View Fn m in . . .. hennatnrvt, .p.tl,YV i.. Products ot .. - 4* metabolism - ) _..!A >>1ha -NT - Conc.Gradients- MassTransfer Effects (Minimal) - "Macroscopic" View - CellularMetabolism Apparent rates of metabolism (experimentally measured- includesmasstransfer effects) - Intrinsic rates (basedpurely on enzyme kinetics) ,LV ~A~ 'channel (Pe >>e (P >> 1) ) "Rate of Metabolism" In the linear regime of Michoelis-Menten kinetics: (AC)* I At n,,, -Vm Figure 5-12: A schematic analysis of the limiting case of high concentration of drug (substrate) added to the 3D culture. Mascuet fTcnlg Massachusetts Institute of Technology :nttt 16 -126- itchooyPoesEgneigCne Biotechnology Process Engineering Center This results of the simple schematic analysis is also borne out by the prediction made by the model developed in Chapter 3 to describe the tissue distribution of any metabolite (In Chapter 3, results were shown for oxygen). The minimum scaled concentration of testosterone anywhere in the tissue is nearly 9 9 % of the inlet substrate concentration (Figure 513), using Scenario 2 modeling analysis. ' .. . I2 1 "..x nrA-* - - U'.UJLsub C,,b + 0.25 ' Co Ub= 200uM 0.996 KM = 5 0 uM 0.996 High Substrate conc.regime 0.994 Testosterone Metabolism 0.992 6Bhydroxylation 0.99 KM 0.988 Figure 5-13: Results for the tissue distribution of testosterone The comprehensive mass transfer model developed in Chapter 3, predicts a negligible tissue concentration gradient at large values of substrate concentration of testosterone (greater than 200 rtM). This result confirms the intuitive schematic analysis of Figure 5-11. 5.6 Summary of key results and conclusions The total internal volume of the reactor and the fluidics was reduced by incorporating a small-volume conical reservoir into the fluidic circuit, instead of the 15 mL reservoir that was used in the original design. In addition, the use of small-bore Teflon® tubing in the fluidics Technology Institute of Massachusetts Massachusetts Institute ofTechnology 127-127- Biotechnology Process Engineering Center Biotechnology Process Engineering Center helped decrease the dead volume in the reactor fluidics. The CFLEX ® pump tubing was coated with a pin-hole free layer of Parylene that helped minimize the adsorption/ absorption/ binding of the drug or xenobiotic to the tubing, to less than 5%. Testosterone at concentrations high enough to saturate the various CYP450 enzymes involved in its biotransformation, was used as a substrate to measure the average rate of formation of many of the hydroxylated products. A method to isolate, detect and quantify the various biotransformation products of testosterone, at high concentrations of substrate that leads to the saturation of the enzymatic biotransformation pathways, was developed. Massachusetts Massachusetts Institute Institute of of Technology Technology -128- -128- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Chapter 6 Comparing Basal Biotransformation Capacity of various in vitro Systems In this chapter, the 3D microfabricated bioreactor that was designed and characterized in the previous chapters is compared with a number of standard in itmrcultures currently used in the pharmaceutical industry. The suitability of the tissue formed in the 3D bioreactor cultures, as an in vitromodel to study metabolism and toxicity of xenobiotics was assessed by comparing the basal expression of the p450's (i.e. Phase I), Phase II genes and genes that regulate the expression of liver-specific proteins, in the 3D bioreactor cultures and in standard in vitro cultures with gene expression profiles seen in ivoand in percolled isolated hepatocytes Using a Rat U34A Affymetrix Chip, genes that are differentially expressed between the 2D collagen sandwich cultures (2DCSW) and a variety of 3D cultures, including the 3D bioreactor (3DB), were identified at multiple time point's up to 20 days in culture, relative to the expression profiles in the in ivo liver. Using RT-PCR, the basal expression of a number different CYP450's and Phase II metabolism genes were compared in seven day old 2DCSW and the 3DB cultures. Since the expression of a number of liver-specific genes are regulated by transcription factors [17, 23, 124-129], the basal expression of these transcription factors were compared between the 2DCSW and 3DB systems, seven days after cell isolation. The expression levels of all these genes in 2D and 3D cultures have been compared to the levels seen in both freshly isolated hepatocytes as well as in in vivo liver. In addition, the activity of the key Phase I enzymes have been compared across the 2DCSW and 3DB liver tissue phenotypes, using testosterone as a candidate substrate. 6.1 Global gene expression profiling of tissue phenotype The global gene expression profiles of the rat genome as represented by the Affymetrix Gene Array (U34A) was compared across isolated cells and tissues formed in 2D - 129 MassachusettsInsfitute of Technology -129- Massachusetts Institute of Tchnology Biotechnology Process Engineering Center~~~~~~~~~~~~~~ Biotechnlolog Pocess Engineering Center collagen sandwich cultures, 3D matrigel spheroids, spinner flask 3D spheroid cultures, and the 3D microreactors. The comparative expression levels of all genes of the culture systems relative to the in vivocells were determined in order to identify major gene classes that differ substantially across the different culture systems. The Affymetrix small sample labeling protocol (www.affymetrix.com) was used for all total RNA samples to generate labeled cRNA. Labeled samples were hybridized in triplicate to the Rat Genome Array (U34A) Gene Chips. Data from arrays were processed using non-linear normalization approach through DNAChip Analyzer version 1.2. The average and the standard deviation of replicates were calculated and probes that were "Absent" throughout the data set were eliminated from further analysis. Fold changes were calculated by dividing the mean of replicates of the culture condition by that of intact liver tissue for each gene. Quality filtering using coefficient of variation (CV) was performed to data with high variability. Using S-Plus software (Mathsoft, Cambridge, MA), a 95% CV quantile was calculated for each sample. Significance was assigned for samples within 95% CV threshold and samples with high variability eliminated. The number of probe sets remaining after filtering was 2098 probes (23.84%). A detailed report on the specific protocols used in the microarray experiment is available elsewhere [130]. The global transcriptional analysis showed that of the -2100 genes that were present following data analysis (see methods), more genes remained unchanged (relative to in tvi) in 3D microreactor culture than in other culture methods at comparable times points (Table 6-1). Of particular interest for pre-clinical drug biotransformation and p450 induction studies, genes that were differentially expressed between the long term 3D microreactor culture and other culture methods include the CYP450's. A hierarchical clustering of the expression of the CYP450s reveals that the 3D microreactor culture system maintains expression levels closer to in vivo liver and isolated hepatocytes than the other culture systems (Figure 6-2). For each culture method, the basal expression levels of CYP450's observed seem stabilized at the two later time point's, and from day 7 forward in the 3D system. Stabilization of basal CYP450 genes by day 7 in 2D collagen sandwich cultures has previously been observed [131] and in house RT-PCR studies (described in the next section) were also used to confirm that expression of several key CYP450 genes were stabilized by day 7 (Figure 6-1). Based on these Massachusetts INassachusetts Institute Institute of of Technology Technology -130- -130- Biotechnology Process Engineering Center Biotechnology Pocess Engineering Center results, the 7 day time point was chosen as a reasonable predictor of long-term differential gene expression among the culture systems, and the gene and protein expression profiles were probed more broadly and quantitatively using RT-PCR and biochemical activity studies. I - FC Base Liver Reference 2D Collagen Sandwich < -4 fold or highly down regulated - - -4 to -2 or | -2 to +2 or moderately unchanged down regulated - - - +2 to +4 or moderately up reaulated +4 to max or highly up regulated Perfused Liver I 4 1950 138 5 Isolated Liver Cells 1 7 2024 61 5 Purified Hepatocytes 8 12 2043 30 5 Day 3 129 177 1330 343 119 Day 13 98 165 1264 357 214 Day 20 291 445 1209 114 39 Day 3 56 108 1567 317 50 Day 13 84 163 1312 336 203 Day 20 89 113 1301 345 250 Day 3 92 120 1285 400 201 Day 7 60 95 1396 404 143 Day 13 64 120 1433 333 148 Day 20 72 145 1335 348 198 cultures Matrigel Cultures Spheroids 3D micro reactors Table 6-1: Global comparison of gene expression profiles across different in vitrocultures at various time points Technology Institute of Massachusetts Institute Massachusetts ofTechnology - 131 131- - Biotechnology Process Enneering Center Biotechnology Process Engineering Center n U w - - == _; i .S e I a I ae 0E Xi aa L I -1l (i) @M * II * * o o Day 3 Day 7 Day 10 Day 14 _- *ft 1A2 2E1 3A2 2C11 Figure 6-1: Stable expression (though downregulated in many cases) of many of the CYP genes is seen in the 2D collagen sandwich cultures after seven days in culture. Technology Massachusetts Massachusetts Institute Institute of of Technology 132 -132- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center LI z 0 <-4 Fold >4 Fold Hierarchical Clustering -- I1 . ...- i { !..............-- __ _ -- I- i I I -I i , .. - I -. Gene Syml Cyp2c13 Cyp2c22 Cyp2d 18 CYP3A9 CYP4F4 CYP4F5 CYP4F6 Cyp2al Cyp2b15 Cyp2c39 Cyp3a 1 Cyp4a10 Cyp4a12 Cyp7al Cyp17 Cyp19 Cyp1bl Cyp27b1 Cyp2a2 Cyp2fl Cyp4bl Cyp51 Cyp7bl Cyp2c Cyp2c2 Cyp2d3 Cyp4f14 Cypl 1b2 Cyp2d5 Cyp2b3 Cyp3A1 CYP3A2 Cyp2E1 -5 &S75 0 Zsc -d -2 I Z-n Figure 6-2: Log ratios of all culture systems (2I), 3D) versus intact liver tissue were clustered for all CYP450 genes as represented on the Affvmetrix Rat Genome (U73A) arrays. Genes Nith higher expression than in intact liver are shown in green, and with lower expression are shown in red. The 2D collagen sandwich culture system (henceforth called "2D culture") was chosen as representative of standardcultures to compare to the 3D microreactorsystem as both the matrigel and collagen gel cultures clustered closely together in dhe p45 0 heat map (Figure 6-2), .asschuscas Instirute of Technoloy - 133- Biotcchnology Process Engineering Center while the 3D microreactor clustered closest to liver (Figure 6-2). However, in order to be able to compare the various in .itro systems quantitatively using the expression of specific genes identified by the global gene expression experiment as a metric, it is important to identify a well-conserved housekeeping gene that does not change across the different systems. More quantitative RT-PCR studies need such a gene to be able to normalize the liver specific gene expression data. 6.2 Quantifying relative gene expression: The two step process: Reverse Transcription - Polymerase Chain Reaction (RT-PCR) In order to validate the'results of the global gene expression data shown in the form of the cluster map in Figure 6-2, more quantitative Reverse Transcriptase-Polymerase Chain Reaction or RT-PCR studies are needed. Real-time RT-PCR is one of the most sensitive methods for quantitation of gene expression levels [132]. This two-step assay measures the accumulation' of fluorescent DNA product during amplification in the polymerase chain reaction (PCR) and correlates this to the initial amount of target RNA. The various steps involved in RT-PCR are detailed in Figure 6-3. First, template RNA is reverse transcribed to i nDNAs cDNA using specific primers, random hexamers, or oligo-dT primers. cDNA is then copied and amplified in PCR to generate double stranded DNA products. s extraction Sample D -l-ll RNA ( r <a 5D SYBH green . PCR, RT Reagents!, Separated RNA (in vivotissue, isolated Analysis cDNA ...... .L_-- L __ heps, and in vitro samples) _ Figure 6-3: The various steps involved in an RT-PCR experiment used to quantify relative gene expression In PCR reactions there are three regions (Figure 6-4): (1) 'baseline' phase -- where primer is in excess and cDNA is limiting; (2) 'exponential growth' phase - where primer and nttt fTcnlg Mascuet assachusetts nstitute of'lechnology ____. _11 Eniern Cete -- 13-BoehooyPoes 134Biotechnology Process nginecring Center _· __ __ cDNA concentration are on the same order of magnitude; and (3) the 'plateau phase' - where cDNA is in excess and primer is limiting. There are currently four competing techniques available that detect amplified products with about the same sensitivity: (1) Molecular beacons; (2) Hybridization probes; (3) Hydrolysis probes - like TaqMan 5'-exonuclease assay; and (4) DNA binding dyes. Each technique has its own unique merits, but DNA binding dyes are relatively cheap and are known to be sensitive enough to detect binding for genes expressed at moderate to high levels in the samples. Figure 6-4: The three regions seen in the fluorescence-cycle number amplification curve obtained following RT-PCR DNA-binding dye detection involves the direct binding of a fluorescent dye (SYBR Green I) into I)NA during PCR. SYBR Green I has an undetectable level of fluorescence when it is in its free form, but each time the cDNA is copied (cycle) the SYBR Green I is bound to a dsDNA (Figure 6-5) and begins emitting fluorescence [132]. SYBR Green * Og 0.0 0................................. . ",,7 -.m' Figure 6-5: Fluorescence following binding of the SYBR Green dye to ds-DNA Technology Institute of Massachusetts Massachusetts Institute of Technology - 135 135- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center When monitored in real-time, this results in an increase in the fluorescence signal that can be observed during the 'exponential growth' phase. In the 'exponential growth' phase a fluorescence signal threshold, C.r (Figure 6-4) is set to determine relative cycle numbers i.e. number of cycles needed to produce a given amount of product that is proportional to the fluorescence signal corresponding to the threshold. This threshold is calculated after normalizing for the global minimum background fluorescence. The value of C,. is directly proportional to the amount of starting template and this forms the basis for calculating rRNA or mRNA expression levels as follows: a(t) PCR,i2c(t) where 'a' is fluorescence intensity, and ' is the efficiency of PCR amplification of target primer 'i' (should be unity). These gene expression levels can be quantified to a resolution of that seen even in a single cell [133]. RT-PCR is thus a highly sensitive, fast, accurate and quantitative means of measuring gene expression compared to other end point assays [134]. Further, the technique is sufficiently high throughput, allowing 96 samples to be run simultaneously in two hours. Quantification using gene expression is prone to variability due to a numbr of technical and other reasons [135-1371. There exists the possibility of co-amplification of genomic DNA and primer-dimers [138]. These unwanted products can be controlled, but require careful primer design (e.g. by choosing primers on the intron-exon boundary and by using primers that result in a product with melting temperatures greater than those of primerdimers and genomic DNA products) and experimental control (by including DNA digestion steps in the protocol and by always having a no-RT control i.e. an mRNA sample that has gone through a DNA digest step but has not been converted to cDNA). Additionally, small pipetting errors propagate into large differences in gene expression levels, so extreme care must be taken to ensure the technical consistency of plate loading for PCR. Furthermore, variations in amplification efficiency have been observed in different primers (in housestudies). -136Institute of Technology Massachusetts Massachusetts Institute of Technology - 136- BiotechnologyProcess EngineeringCenter~~~~~~~~~ Biotechnology Process Engineering Center Thus, each primer must be validated to ensure a linear trend in the concentration or amount of gene product and threshold cycle number (keeping fluorescence constant or vice-versa). Figure 6-6 shows the linear relationship obtained between the threshold cycle number at constant fluorescence versus the amount of 18s gene present (represented here as absolute cell numbers assuming that the amount of 18s gene per cell is constant). 4A I L+ y = -1.5617Ln(x)+19.043 ) 10e3 I- Go dI! 100 1000 Cell Number (1000s) Figure 6-6: Linear relationship between threshold cycle number and quantity of gene product for the 18s gene. The above plot (mean+SEM, n=18) was generated by combining a known volume of concentrated cDNA in a real-time RT-PCR reaction. Theoretically, the slope of the line should have been 1/ln(2) = 1.4, as we should ideally expect twice the amount of product (18s gene) per unit change in Cl value. However, in reality primer binding efficiency as well as the efficiency of binding of the DNA dye, may not be 100% leading to higher values of the slope (and hence less than two-fold change in product amount per unit change in threshold cycle number for the same fluorescence). A detailed analysis of RT-PCR protocols, techniques and methods is available elsewhere [54]. Technology Massachusetts Massachusetts Institute Institute of of Technology 137-137- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 6.3 Identification of a well-conserved housekeeping gene for RT-PCR studies Normalizing mRNA expression level data of various genes is of critical importance when comparing systems containing different cell numbers as well as cells that have been under different culture environments, as extraction efficiencies may not be equal in all systems. In addition, the normalization can help differentiate between technical errors associated with performing the assay and actual differences in gene expression that can then be ascribed to differences in the culture paradigm or cell treatment methods used. It is necessary to find a gene whose expression is equally conserved across all cell culture systems being investigated, as well as in the presence of any toxin or drug added to the system. This latter is an important consideration that needs to be bome in mind if data on enzyme biochemical activities of specific p450's are to be used to compare the various in itm systems.In addition, data on changes in the expression of specific p450's in response to the addition of any drug or inducer will need a well-conserved gene that is unaffected by the addition of the drug/ inducer, to normalize the data. Previous studies have used total DNA, GAPDH, P3-Actin,total RNA, and ribosomal RNA (e.g. 18s and 28s) as a means to normalize data [135, 139]. Certain studies have expressed concern over the consistency of these expression levels between various in vitro cell culture systems as well as repeatability within the same culture system [140, 141]. DNA is degraded slowly in comparison to RNA and may be indicative of both live and dead cell densities. Previous studies, as well as in house data in our lab [130] have suggested that mRNA expression levels for GAPDH may not be equally conserved [142]. Expression levels for 3-Actin may be significantly different in 2D and 3D cell culture systems because of variations in structural complexity between 2D and 3D in vitro culture systems and this is indeed seen to be the case (Figure 6-3). Total RNA levels are thought to be largely a function of cell number, but cannot be used to normalize real-time RT-PCR data because of differences in cDNA conversion efficiencies between samples. Ribosomal RNA makes up 85-90% of total RNA and we hypothesize that it also scales linearly with cell number (Figure 6-6 validates our hypothesis). These methods of normalizing data must be evaluated to accurately validate experimental data. It may also be possible to determine the approximate the number of cells in a sample using either total RNA or critical threshold (C.,) values for 18s expression from Massachusetts Nbssachusetts Institute Institute of of Technology echnology - 138- -138- Biotechnology Process Engineering Center Biotechnology Process Engineering Center real-time RT-PCR in combination with a standard curve of known RNA concentrations. Figure 6-7 plots the linear relationship obtained between percolled isolated hepatocyte cell numbers and the total RNA isolated. Together with Figure 6-6, this proves that our hypothesis - namely that the amount of 18s RNA per cell is constant and that the total amount of 18s RNA as a fraction of the total isolated RNA is constant. y=0.065x R2 =0.98 0) = 0 I- 1 0 100 200 300 400 500 600 700 800 Cell Number (1000s) Figure 6-7: Isolated Hepatocytes vs. Total RNA A Beckman Coulter DU 460 spectrophotometer was used to measure absorbance at 260nm and to determine total RNA levels in isolated hepatocyte samples containing various cell numbers (10IK-800K, n=47). Total RNA was plotted (mean+SEM) and shown to be a linear function of cell number. For the chosen housekeeping gene to be used to normalize gene expression data from different in vitr culture systems, specifically from different 2D and 3D tissue culture systems, it is important that the amount of the housekeeping gene per cell be invariant across the different culture systems. RT-PCR reactions against the 18s gene were performed on equal - 139Insdtuteof Technology Massachusetts - 139Massachusetts Institute ofTechnology Center~~~~ Biotechnology Process En~~~~~~~~~~~~~~~~~~~~~~~neering Biotechnology Process Engineering Center quantities of total RNA isolated from 2D and 3D culture. From Figure 6-8, where the C,. value is indicative of the number of cycles needed for a given amount of 18s DNA product (given that all three systems started off with the same amount of total RNA), it can be seen that the amount per cell of thel 8s gene is statistically invariant across the different culture systems. 15. 10- 5- n. v Isolated Hepatocytes 2D Cultures 3D Bioreactors Figure 6-8: 18s gene per cell is invariant across the different culture systems. Real-time RT-PCR was used to determine C.. values for freshly isolated hepatocytes (13.93+0.20, CV=0.07, n=24), for 2D collagen sandwich cultures (13.22+0.10, CV=0.05, n=42), and the 3D liver bioreactor (13.98±0.08, n=3). The coefficient of variance of C. values across all samples was 0.06 or 6%. 2D collagen sandwich cultures and the 3D bioreactor had the same level of 18s expression for the same amount of total RNA isolated from the three different systems, as freshly isolated hepatocytes as determined by the Bonferroni Method. Plot shown above is mean+SEM. Figure 6-9, shows the effect of addition of a drug testosterone on the expression of 18s in 2D collagen sandwich cultures. Testosterone is an important substrate used in quantifying the biochemical activity of a number of pharmaceutically relevant p450's. RT-PCR reactions against the 18s gene were performed on equal quantities of total RNA isolated from different 2D cultures taken down after being exposed to the same concentration of drug (250 pM) for different intervals of time. From Figure 6-9, where the Cr value is indicative of the number of cycles needed for a given amount of 18s DNA product (given that all the samples 1 ~ Inttt Massachusetts~~ ~ ~ ~ ~ ~-140- Massachusetts Institute ofTechnology itcnloyPoesEgneigCne DfTcnlg Biotechnology Process Engineering Center started off with the same amount of total RNA), it can be seen that the amount per cell of thel8s gene is indeed statistically invariant across the different samples and is therefore not affected by the addition of testosterone as a candidate drug. 15- -7 10- 0I5- ._L nV- _ 15 30 45 60 90 120 Time after testosterone addition (min.) Figure 6-9: Effect of addition of testosterone on the per cell 18s expression levels in 2D collagen sandwich cultures There was no statistical difference in the C. values across the different cultures taken down at different time points after administration of the drug (n= 18 at each time point) 6.4 Analysis of RT-PCR data: Use of normalized fold change as a metric to compare gene expression data There are two primary methods used to quantify gene expression levels: absolute and relative quantification. The former method finds a lot of use in literature [132, 134, 135, 138, 139, 143]. The mathematics for the use of the most generalized expressions needed to quantify the relative expression of a particular gene, following normalization with a chosen housekeeping gene, was also developed as part of this thesis. This is described in detail in subsection 2 of this section. Technology ofTechnology Massachusetts Institute Massachusetts Institute of - 141 141- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center 1)Absolute Quantification In this method of quantification, samples of known concentrations of gene products are used to construct a standard curve that relates the C.1 value with the concentration of the gene in a sample. Logarithmic regression is used to fit slope, m, and the intercept, b, into the form: CT=mln(CA)+b (6-1) Thus the absolute unknown concentration of a particular gene A in a sample, may be calculated using the logarithmic relationship and the calculated value of C. from the RT-PCR amplification curve. 2) Relative Quantification Relative quantification involves reporting the expression of a gene A in sample 'a' relative to the expression level the same gene A in another sample 'b'. The difference in the value of the threshold cycle numbers corresponding to the same amount of fluorescent gene product, quantifies the relative up-regulation or downregulation of the gene A in sample 'a' relativeto 'b' or vice-versa. Whether the data is reported in absolute terms or in relative terms it is important to normalize the data using relative expression of a house keeping gene (in our case 18s) between the two samples. The mathematical relationship that determines the expression of gene A in sample 'a' relative to the expression of the same gene A in sample 'b', is derived as below: Let the concentration of gene A in sample 'a' be CA, that of the 18s housekeeping gene in sample 'a' be CB8s . Let the concentration of gene A in sample 'b' be CA and that of the 18s housekeeping gene in sample 'b' be Cb8 . From equation (6-1-) we have: Massachusetts Massachusetts Institute Institute of of Technology Technology 142 -142- Biotechnology Process Engineering Center Biotechnology Process Engineerig Center CTa = mA n (Ca) + bA (6-2) n (Cb) + b CTAb =m (6-3) Subtracting (6-3) from (6-2): C C;,a -C T,bn= In C aA A A (6-4) Similarly for the housekeeping gene 18s, we have: 18s O18s CT,a CT,b n ( C a s8s m18 iC (6-5) Cb) Subtracting (6-4) from (6-5): / II A (C185 S (c8s -- Ta CA T,a - s _ Tb) Ab)=lnn T, -8s (6-6) Cb2) t bA (C,18s kT*a - i.e. where l [(FC ) 18s in 'a' over A T,a II (C18s CA \ T,h T,b (6-7) is the Fold change in 'a' over 'b' of 18s and the [(FCb)Auis fold change ' of gene A. - 143 Institute of Technology Massachusetts - 143Massachusetts Institute of Technology Biotechnology Process Engineering Center~~~~~~~~~~~~~~~~~~~~~~ Biotechnology Process Engineering Center Thus T ) FC(C) 8 ba TCb j) (6-8) Theoretically, for a unit change in the value of CT and CT , the concentration of the 18s gene and gene A should respectively double. This will mean that the theoretical values of: Thus mA = ) = mss ( (6-9) In reality, a unit change in C. may result in a less than two fold increase in the amount of gene product. Hence the value of ml and m,, may be higher than that suggested in (6-9). Also, a closed form solution and simplification for equation (6-8) is only possibly by the theoretical assumption made in (6-9). Using (6-9) in (6-8), this simplifies equation (6-8) to: =,CTa Tb-CTb)2 C 7 (6-10) Thus we can define a relative normalized fold change of gene A in sample 'a' over sample'b', normalized using the expression of the 18s housekeeping gene as: Normalized Fold Change (NFC) of A in sample 'a' over sample 'b' is given by: (NFC )-= 2 (CT'a CTa Y(T , b )) (6-11) The normalized log fold change (LNFC) of sample 'a' over sample 'b' in given by: Massachusetts Miassachusetts Institute Institute of of Technology Technology -144-144- Biotechnology Process Engineering Center Biotechnology Process Engineering Center (LNFC)= {{CA _18s log 2 (2((Ca -c,CT \ - A bb 18s ) (6-12) Figure 6-10 shows the log fold change in the expression of 18s and L-actin relative to the expression in isolated hepatocytes, 2D collagen sandwich culture and 3D bioreactor 18s vs. P -Actin Normalization 6 N=6, n=6 n C . 0) LIu O n mRNA ,_1 LL lagen 0) -j ich Culture Figure 6-10: 18s versus b-actin as a normalization gene P-actin, a commonly used housekeeping gene is seen to be upregulated in 3D cultures relative to in cicoand is seen to be downregulated (again relative to in tviv) in the 2D cultures. On the other hand 18s is seen to be a very stable housekeeping gene whose expression amount per cell is seen to be invariant across 2D and 3D cell cultures. 6.5 Comparing liver specific gene expression between in vitro cultures using Affymetrix® microarrayand RT-PCR studies As described in Section 6.1, an initial assessment of the relative basal metabolic capacities of different culture systems compared to rat liver was obtained by analyzing the global gene expression profiles of the rat genome as represented by the Affymetrix Gene Array Masachsets nsitue MassachusettsInstituteof Tchnology o Tchnloy -145-14 - iotcholoy rocss ngneein Cete Biotechnology Process Engineering Center (U34A). A hierarchical clustering of the expression of the 24 significant CYP450s revealed that the 3D microreactor culture system maintained expression levels closer to in vim liver and isolated hepatocytes than either matrigel or collagen gel sandwich cultures after 7, 13 or 20 days of culture (Figure 6-1). At day 20, most CYP450's were strongly downregulated og2 fold change < -4) in collagen gel sandwich culture and matrigel culture (15-16 genes of 24), while only a few (5 genes) were strongly downregulated in 3D microreactor culture (Figure 6-11). The basal expression levels of CYP450's appeared to be stabilized after a week in culture, consistent with what has been previously observed in 2D collagen sandwich cultures [131]. Westerns blots (Figure 4-10) and RT-PCR studies (Figure 6-1) confirm that expression of several key CYP450 genes were stabilized perhaps by day 3 and certainly by day 7 in collagen gel sandwich cultures and 3D microreactor cultures. Based on these results, the 7 day time point was chosen as a reasonable predictor of long-term differential gene expression among the culture systems, and the gene expression was probed more quantitatively using RTPCR and biochemical activity studies. The 2D collagen sandwich culture system was chosen as representative of standard cultures to compare to the 3D microreactor system to, as both the matrigel and collagen gel cultures clustered closely together in hierarchical clustering, while the 3D microreactor clustered closest to liver. Massachusetts Massachusetts Institute Institute of of Technology Technology - 146 -146- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 16 14 U) C 12 0) o 10 I) :- 8 L 6 E4 . Z 2 min to -4 -4 to -2 to +2 +2 to +4 +4 to max Log2 fold change relative to native tissue 1I 16 14 S 12 a) A 10 Ve 8 0 0. 0 L. 6 Z. z 2 0 min to -4 -4 to -2. -2 to +2 +2 to +4 .+4to max Log 2 fold change relative to native liver tissue Figure 6-11: Relative CYP450 gene expression across cultures - from RatU34A microarray Massachusetts Miassachusetts Institute nstitute of of Technology Technology 147 -147- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center The CYP450 enzymes are diverse in their expression and activity levels, substrate specificities, and regulatory mechanisms. For quantitative analysis by RT-PCR, a subset of nine CYP450 genes were chosen that reflect this diversity and that have previously been studied in vivo and in vitro under a variety of conditions and are of relevance in pharmaceutical development. Similar criteria were used for selecting Phase II genes, with an emphasis on genes that have previously been reported as down regulated in vitro. The expression patterns in the 3D microreactor and 2D culture were strikingly different after 7 days in culture (Figure 6-12). Whereas all Phase II genes and almost all Phase I genes (7 of 9) were strongly down-regulated in 2D, most genes in 3D culture are either unchanged or slightly down regulated (Figure 6-12). CYP 2E1 was the only gene that approached the category of being strongly downregulated in 3D culture, and its expression level was about 30-fold higher in 3D microreactor culture than in 2D culture. CYP 2E1 gene expression is reportedly down-regulated by insulin [144] and is hence expected to be down regulated in a culture responding physiologically. With the exception of CYP 1A2, which tends to be stably expressed in most cultures [80] and is slightly upregulated in both 2D and 3D cultures here, loss of CYP expression in primary rat hepatocyte cultures has been documented in a number of different culture configurations [8, 12, 65, 80]. CYP 2C1 is a major constitutively-expressed CYP450 in male rat liver and is particularly susceptible to changes in the hepatocyte environment [120]. The early loss of CYP protein activity in primary hepatocyte cultures has been attributed to the activation of nitric oxide synthesis, due to the generation of reactive oxygen species during the isolation procedure [80, 145], and may involve other mechanisms as well [146]. Formation of three dimensional multicellular cell aggregates may mitigate oxidative-stress induced tissue damage [80]. Indeed, 2C11 expression is observed to be only slightly down-regulated in the 3D culture but is strongly downregulated in 2D culture (Figure 6-12). The expression of CYP2B1/2 is moderately upregulated in 3D microreactor cultures (Figure 6-12), for reasons that are as yet unknown. Induction by agents leached from components of the 3D microreactor system as one possible reason for this upregulation was ruled out - 2D cultures that were cultured up to 7 days with conditioned medium circulated in the 3D microreactor system did not show any Technology Massachusetts Massachusetts Institute Institute of ofTechnology 148 -148- Biotechnology Process Engineering Center Biotechnology Process Egineering Center '~.~~ 6 · - "i i o1i ' i U) C\I : ~ .o to UeU~~ U, 0 _ _ _ _ _ _ _ _ _ U ~UUU _ 0 N oUo .n CC i U ~.~ U.: v-1 4 4- U N I 4 cd QU .4 -O -J -e o · ~ ~~~ 0 LO o~ao ~ U~-- s'- - c 'J oo i 44 CL4 -Z CI U0, U-ell OD 0 0 cm O C- O _ ii u 0 Ir t..BXgtol O5:c4 QJ 0 &J ' 6 UU U mOCF 4U ,,d~ e. (O!A u! ol eAllela a6OueI4)plo) Z601 Massachusetts Institute ofTechnology - 149- Biotechnology Process Engineering Center upregulation of 2B1/2 relative to control (data not shown). CYP 2B1/2 shows a zonal activity gradient in vivo [42, 147], with more activity in the pericentral zone of the hepatic acinus, and thus the environment in the microreactor may reflect these conditions. Among the Phase II genes, downregulation of some of the UGT's, GST-Ya and specifically that of HSST to lower than 20% of the in i values within 24 hr after cell isolation has been reported in a variety of primary cultures, independent of the culture matrices and media formulation used [148]. However, the expression of these genes, along with that of UGT-1A, is maintained at near in vivolevels in the 3D microreactor tissue. 6.6 Relative expression of liver transcription factors between 2D and 3D cultures Liver-specific gene expression is believed to be controlled primarily at the level of gene transcription [128, 129] and the expression of drug metabolizing genes are regulated by key liver transcription factors that play a major role in the overall maintenance of the differentiated state of hepatocytes [23, 124, 126]. Expression of HNF's, a heterogeneous class of evolutionarily conserved transcription factors that contain several families of liver-enriched transcription factors required for cellular differentiation and metabolism, is strikingly different in 2D and 3D culture, with most factors unchanged in 3D culture and downregulated in 2D culture (Figure 6-13). HNF-4a is an upstream regulator of HNF-1 a expression [149] and both HNFla and HNF4a, are regarded as important regulators of the transcription network in liver-specific gene expression [124]. HNFla and HNF13 are both down regulated in isolated hepatocytes, but show recovery in 3D over 7 days of culture and sustained downregulation in 2D culture. The strong downregulation in HNFa at day 7 in 2D culture is consistent with the moderate downregulation of HNF4a seen in these cultures (Figure 6-13). The expression of CEBP-[, is upregulated under all conditions, with by far the strongest upregulation observed in 2D. Over expression of CEBP-P has been reported in literature in response to tissue injury and stress [150]. The 3D perfused microreactor may help mitigate the effect of the isolation stress more effectively than the 2D environment. The transcription factor expression profiles for the two culture conditions are thus consistent with the expression profiles of the downstream Phase I and Phase II enzymes, and are further consistent with differences in the Technology Institute of Massachusetts Mlassachusetts Institute ofTechnology -150-150- Biotechnology Process Engineering Center Biotechnology Process Engineering Center albumin secretion rates previously observed in the 3D microreactor system compared to other primary rat hepatocyte culture systems [47] .g tr. 0 C. 0 la -s Figure 6-13: Basal expression of hepatic transcription factors in isolated hepatocytes, 2D collagen sandwich culture (day 7), and 3D microreactor (day 7) cultures expressed as log2 -fold change relative to liver in vie. 6.7 Measurement of testosterone metabolism in 2DCSW and 3DB cultures The activity of the Phase I CYP450 enzymes measured by the rates of testosterone hydroxylation to products 16a, 16p, 6P hydroxytestosterone parallels the differences seen at the mRNA level (Table 6-2). The basal 16a hydroxylation rates, a measure of CYP 2C11 activity, are about 8-fold higher in 3D culture compared to 2D, a difference corresponding to the -8-fold greater 2C11 mRNA level observed in 3D microreactor culture compared to 2D culture (Figure 6-12). The combined activities of CYP2B1 and CYP2B2 measured by rate of production of the 16[ hydroxylation product [106, 118] of testosterone are nearly 20 fold Technology Massachusetts Massachusetts Institule Instituie of ofTechnology 151 -151 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center higher in 3D cultures than in 2D (Table 6-2), a result that parallels the trend but is muted in terms of absolute magnitude of the -200-fold greater expression observed in RT-PCR analysis of CYP2B1 mRNA levels. These differences in magnitude underscore the importance of validating results observed by PCR with activity data, as activity depends on a multitude of downstream events including mRNA stability and translation, protein stability, and co-factors needed for protein activity. For example, nitric oxide and/ or peroxynitrites induced during isolation can cause specific blockage of the hemes of the CYP450s, resulting in alteration of enzyme activities in ittv through mechanisms downstream of mRNA [151]. Among the several CYP450s that have been reported to hydroxylate testosterone to form 6P hydroxytestosterone, CYP3A1 and CYP3A2 , perhaps along with CYP1A2, appear to dominate the activity [120, 146, 152]. The 20-fold difference between 2D and 3D microreactor cultures in the combined activity of enzymes that contribute to formation of 63 products trends with the -100-fold difference in CYP3A1/3A2 mRNA levels (Figure 6-12), as well as with differences in the message levels of some minor enzymes that might contribute (1A2, 2C11). Testosterone hydroxylation rates reported in the literature cover a wide range, reflecting variations due to differences in animal strain (different strains have different basal expression rates), measurement protocols (liver microsome fractions compared to whole cells), and methods for normalization to obtain a specific rate per cell [120, 146, 152]. With these considerations in mind, our rates of metabolite formation in the 3D bioreactor are reasonably comparable to those for freshly-isolated cells or in vivorates, while the metabolism rates in 2D culture are generally substantially below or at the low end of the reported rates (Table 6-2). . Also, in normalizing the activity data, the total number of viable cells were calculated based on a measurement of the total 18s rRNA content (see Section 6.4, 6.5, Figure 6-6 or 6-7 may be used to back out a 'viable' cell number, see Appendix for RNA isolation, cDNA preparation and RT-PCR protocols), while most reports in literature use total cell number from total DNA measurements that may account for both live as well as dead cells. :nttt Masacustt fTcnlg Massachusetts Institute ofTechnology 12 - 152- itcnlg wesEgneigCne Biotechnology Process Engineering Center Testosterone Hydroxylation Product 16a CYP450's Testosterone Hydroxylation rates on Day 7 mediating basal (pmoles/10 6 cells/min) metabolism' 2C11 2D collagen 3D Other systems (in viv or gelb microreactorb isolated cells) 100 ± 42 840 + 206 850 + 60 to 1880 500' 825 to 1200 d 16P 6j3 2B1, 2B2 Major:3A1, 3A2, 2A2 Minor: A1A1, 1A2, 2C11 49 + 22 878+ 271 145 + 57 2970 +1620 15 to 100' 2700 + 330 to 3500 + 790f 500 to1250d' 6000g Some variability has been reported depending on species and measurement method (i.e., microsomes, isolated cells, or in vivo). Enzymes shown are those reported as primary metabolizers in the most relevant experimental system for which data is available (primary Fisher hepatocytes for 16a27 ; in vivo Sprague Dawley for 1644; Sprague Dawley microsomes for 6[39'though several other enzymes have been suggested as involved in other studies). bValues represent the mean + standard error for N = 4 biological replicates (i.e., different rats) with at least 3 replicates within each animal. c Basal rates for freshly isolated male Fisher rat hepatocytes, this work, range reflects measurements taken after either 15 min (1880 pmoles/10 6 cells/min) or 30 min (850 pmoles/10 6 cells/min) incubation with testosterone; the decreasing rate with time reflects the expected rapid loss of CYP450 activity in suspension culture. d Basal rates for freshly isolated male Fisher rat hepatocytes - ref. [120] Basal rates for Sprague Dawley rats in ivo - ref. [152] f Basal rates for freshly isolated male Fisher rat hepatocytes, this work, range reflects measurements taken after either 15 min (3500 pmoles/106 cells/min) or 30 min (2700 pmoles/ 106 cells/min) incubation with testosterone. gDexamethasone-induced rates for Sprague Dawley rats in vivo - ref. [152] a Table 6-2: Higher basal activity of 2C11, 2B1, 2B2 and 3A1, 3A2 seen in the 3D microreactors over the 2D collagen sandwich cultures. 16[3 and 16a hydroxylation of testosterone, indicative of the activity of CYP2B1/2 and 2C11, are seen to be at least 10 fold higher in 3D cultures over 2D. The rate of 6[ hydroxylation is nsitteofTehnloy Masahuets MZassachusettsInstitute of Technology 13 - 153- BotcholgyPrces ngnerig ene Biotechnology Process Engineering Center seen to be 20-fold higher in 3D over 2D and is even higher than rates seen in liver microsomes. Since multiple p450's including CYP2A and CYP2B, have been reported to contribute to the 613 hydroxylation of testosterone [106, 118], the observed upregulation of CYP2B may also be contributing to this result. In normalizing the activity data, we calculate the total number of viable cells based on a measurement of the total 18s rRNA content. Data from N=3 different rats, with n=3 different replicates in each experiment 6.8 Biochemical regulation of tissue function: effect of soluble factors in the media The biochemical regulation of the various biophysical processes that take place during tissue morphogenesis has been well documented in detailed reviews in literature [8, 50, 88]. Thus, in addition to providing a 3D environment that mimics the biophysical process of in vivo like morphogenesis through cell-cell, cell-matrix, soluble and shear mediated signalling mechanisms, a suitable media formulation with appropriate factors that help mimic the in vivo biochemistry can further improve the tissue phenotype. For e.g. both in 2D collagen sandwich cultures as well as in 3D bioreactor cultures, relative to the in ivogene expression, the bile acid transporter genes are seen to be highly down regulated (see Figure 6-13), possibly due to the absence of Hepatocyte Growth Factor (HGF) in the media [21, 22, 153]. HGF is needed for the maturation and function of the bile duct cells that carry many of the bile acid transporters. Massachusetts MlassachusettsInstitute Institute of of Technology echnology -154- -154- Biotechnology Process Engineering Center Biotechnoloy Process Engineering Center Figure 6-14: Downregulation of Bile and Fatty Acid transporters seen in both 2D as well as in 3D reactors. It is hypothesized that this downregulation may in whole or part be mediated by the presence or absence of specific components in the media used to culture the cells Similarly, insulin present at a high concentration in the media is known to downregulate the expression of CYP2E1 and PEPCK (Phosphoenolpyruvate Carboxykinase) in culture [144, 154-158]. This is seen in the downregulated expression of these genes in both 2D and 3D cultures (see Figure 6-14). The presence of Dexamethasone in the media may result in the upregulation of common genes such as Tyrosine Amino transferase (TAT) and Tyrosine 2-3 dioxygenase (TO) (Figure 6-15). Technology Massachusetts MassachusettsInstitute Institute of of Technology 155 -155- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Figure 6-15: Other genes modulated by specific biochemical components in the media PEPCK and 2E1 are downregulated by insulin, while TAT and TO are marginally induced by Dexamethasone. Catalase is known to be affected and downregulated by peroxynitrites induced during the isolation of the primary cells. 6.9 Summary of key results and conclusions Using a broad spectrum of gene expression and biochemical activity metrics, the cell phenotype during extended culture was seen to be substantially closer to that of liver in the 3D microreactor compared to other traditional in vitroculture methods used in the Pharmaceutical industry, particularly the 2D collagen sandwich cultures. Global transcriptional profiling was first used to identify genes that are differentially expressed in the various culture formats at multiple time points up to 20 days in culture, relative to the liver expression profiles in vio. From these studies a specific time point to compare the basal expression levels of several important CYP450 and Phase II metabolism genes was chosen. In addition, the basal expression levels of transcription factors that regulate broad programs of liver-specific genes was compared across various in itro culture systems, more quantitatively via RT PCR. It is Massachusetts Massachusetts Institute Institute of of Technology Technology 156-156- Biotechnology Process Engineering Center Biotechnology Process Engineering Center hypothesized that the better preservation of the basal expression of the hepatic transcription factors may possibly be responsible for more in vivolike expression of the liver specific genes in the 3D microreactor than in the 2D collagen sandwich cultures. Genes that are likely regulated by soluble factors present in the media were down-regulated in both the 2D collagen sandwich as well as the 3D microreactor cultures. Such genes include the CYP2E1 (regulated by insulin in the media), and specific transporters - the Bile Salt Export Pump (BSEP), the organix anion transporter (OATP1 and OATP2), and the nulti drug resistance gene (MDR2). It is hypothesized that the addition of specific biochemical stimuli such as HGF may help preserve the expression of the specific transporter genes that are present specifically in the bile duct cells. Institute of Massachusetts Massachusetts Instiute of Technology Technology 157 - 157- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Chapter 7 Inducibility as a QuantitativeFunctional Response Marker While maintenance of basal expression of CYP450 is important for the use of in vitt systems as predictive screens for mechanistic toxicity studies, inducibility of CYP450's by drug like compounds is used as an important yardstick to fail new chemical entities (NCE's) during pre-clinical testing [3, 50, 65, 159]. A characteristic of a subset of enzymes of the P450 super family able to metabolize xenobiotic compounds, is their relatively low basal expression in the absence of substrate and their highly elevated expression in the presence of their own substrates or other inducer compounds. In particular, members of the CYP1A, CYP2B, CYP2C, CYP3A, and CYP4A gene subfamilies are known to be highly inducible by certain prototypical xenobiotics. This xenobiotic induction usually is tissue-specific, rapid, dosedependent, and reversible upon removal of the inducer [50, 131, 160, 161]. Most in itm tissue models of the liver are known to lose their potential to be induced by common xenobiotics that elicit an induction response in vivo. Thus, inducibility of CYP450's may be used as a quantitative, fundamental assay to assess the phenotype of a tissue culture model. We compared the effect of addition of prototypical inducers to 2D Collagen Sandwich cultures (2DCSW) and 3D Bioreactor (3DB) cultures. 2D collagen sandwich cultures and 3D bioreactor cultures were set up according to standard protocols described in the Appendix. Prototypical inducers were added four days after cell isolation, and were added to fresh media each day until day 7. Media in both the 2DCSW and 3DB cultures were changed everyday and received the same concentration of inducer. The concentrations of all the inducers used were optimized for maximal induction in 2D collagen sandwich cultures (basedon experimentsdonein the Tannenbaum Lab, by Dr.Kevin Leach). Technology Massachusetts Institute Massachusetts Institute of ofTechnology 158-158- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 7.1 The cue-signal-response hypothesis The induction phenomenon was first recognized because they produced alterations in pharmacological responses to drugs or other xenobiotics. Inducers are often substrates to the induced enzymes; thus, enzyme activity increases only as needed. Second, enzyme induction often increases detoxification, particularly when low to moderate concentrations of substrate are present; thus under most conditions induction is a protective mechanism, whereby the cell can detoxify lipophilic compounds that might otherwise accumulate [162]. Both characteristics are likely to facilitate the survival of the cell in a potentially toxic chemical environment. Induction of CYP450 enzyme activity can be disadvantageous in some instances. For example, the induced enzymes often metabolize numerous substrates; thus enzyme induction by one chemical may lead to increased metabolism of other compounds; if one happens to be a drug of low therapeutic index, the increase in metabolism may lead to the loss of efficacy [163]. Occasionally, enzyme induction may enhance chemical toxicity. For example, high concentrations of the analgesic acetaminophen saturate detoxification pathways, leading to reactions via cytochrome p 4 5 0 that generate reactive electrophiles, which bind to cellular macromolecules and produce hepatic necrosis. CYP450 induction increases the severity of acetaminophen toxicity by increasing the production of reactive metabolites and accentuating the imbalance between detoxification and activation pathways for the drug [164]. Understanding the molecular basis for the induction effect of small molecules, drugs, environmental toxicants on the cellular phenotype, can help provide insights into gene regulation of relatively broad interest. Typical changes seen in cellular phenotype as a result of xenobiotic induction is the enhanced transcription of specific genes, including the CYP450's. Great efforts have been made to decipher the molecular mechanism involved in the CYP induction. Recent studies suggest that major classes of CYP genes are selectively regulated by certain ligand-activated nuclear receptors (NR) [165-167]. The nuclear receptors are a superfamily of genes coding for transcription factors that transform extracellular and intracellular signals into cellular responses by triggering the transcription of NR target genes. Nuclear receptors contain a DNA-binding domain and a ligand-binding domain, which is similar to those classical steroid- and thyroid-hormone receptors [168]. The response element Massachusetts Massachusetts Ins6tute Institute of of Technology echnology -159- -159- Biotechnology Process Engineering Center Biotechnology Process Engineering Center of a nuclear receptor is usually composed of two half-sites related to the hexamer AGGTCA. The nuclear receptor superfamily also contains receptors for non-steroid ligands such as retinoid acid, prostaglandins and fatty acids and so-called orphan receptors, for which no physiologically relevant activators or ligands are yet known. In eukaryotes, DNA molecules are tightly wrapped up by histone proteins forming a structural unit called chromatin. Transcriptional activation of eukaryotic genes usually involves in acetylation of histone proteins sequentially resulting in alteration in chromatin structure. According to the current model of nuclear receptor activation, ligand binding induces significant structural changes in the folding of the ligand binding domain of NR to form a hydrophobic patch that is accessible to common co-activators and co-integrators [168]. Co-activators either possess intrinsic histone acetyltransferase activity or recruit additional histone acetyltransferases that relieve suppressive effects of the chromatin to activate transcription. Although some nuclear receptors show certain differences in the transcription process, transcriptional activation by nuclear receptors usually includes ligand-dependent dimerization of receptors, interaction between transcription factors and a xenobiotic responsive enhancer, transmission of the induction signal from the enhancer to a promoter and changes in chromatin structure. In some cases, protein phosphorylation and dephosphorylation are also involved in the transcriptional activation by nuclear receptors. Different CYP genes are regulated by different transcriptional factors [169-171]. Liver-predominant expression of CYP is driven by distinctive liver-enriched transcription factors. For example, inducible expression of CYP3A is regulated by ligandactivated nuclear receptor CAR (constitutively active receptor) and can be activated by a wide variety of antibiotics, barbiturates and other drugs such as glucocorticoids [165, 166, 172]. For experiments on induction of rat CYP3A's, done as part of this thesis, a non-glucocorticoid 16a Pregnelone Carbonitrile was used as the inducer. A complete list of prototypical inducers of rat CYP450's as well as the primary ligand binding nuclear receptors/ transcription factors involved in the induction pathway are listed in Table 7-1. In the case of CYP1A isoforms, induction is through the binding of a ligand to the aryl-hydrocarbon receptor (AhR) [170], translocation of the ligand-bound AhR into the nucleus, and association of AhR with its dimerization partner AhR nuclear translocator (Arnt). The AhR-Arnt complex then binds to Inttt ofTcnlg Massachusetts Massachusetts Institute of Technology 10 -160- itcnloyPoesEgneigCne - Biotechnology Process Eineering Center xenobiotic response element and turns on the CYP1A gene transcription. Similarly, for CYP4A, induction is through the binding of a ligand, in this case, clofibric acid to the (Peroxisome Proliferation Activation Receptor) PPAR-a nuclear receptor, followed by the translocation of the NR to the nucleus, and association of the NR with the retinoid receptor RXRa. In this case, the dimer binds to the PPRE (peroxisome proliferator response element) present on the CYP4A1 gene. For the induction of CYP3A the nuclear receptor involved is the Constitutively Active Receptor (CAR). Detailed description of the signalling cascade mechanisms involved in the induction of specific CYP's studied as part of the experimental protocol are discussed in specific sections of this chapter. Although CYP induction is normally the consequence of an increase in gene transcription, some none-transcriptional mechanisms also have been reported to be involved [170]. For example, human CYP3A4 induction by troleandomycin appears to be nontranscriptional. Troleandomycin does not activate CYP3A4 gene transcription, but increases enzymatic activity by preventing CYP3A4 proteins from degradation [162]. Similarly,induction of human CYP2E1 by ethanol and isoniazid is not transcriptional, but it results either from protein stabilization or increased protein translation [173]. In this study, 3-Methyl Cholanthrene, 16a Pregnelone Carbonitrile, and Clofibric Acid were used as prototypical inducers to compare the response to 2D and 3D cultures. The mechanism of action and regulation of the effect of these inducers have been well-studied and characterized in both in tivo and in vitmro systems [67, 174-182]. Separate experiments on 2D collagen sandwich cultures were used to determine inducer concentrations that produced maximal induction in 2D collagen sandwich cultures (Dr.KevinLeach, TannenbaumLab: seeFigure 7-3 for an exampledoseresponse).Inducers were added at the same concentration to both 2D as well as 3D cultures. This helped obviate issues of mass transfer effects of inducers on the tissue phenotypic response in 3D. At the same time, reported literature data were used to verify that the chosen concentrations of the inducers were non-toxic to cultures. For e.g. from Fugure 7-3, it is clear than a 10 ±Mconcentration of PCN produces a maximal induction in 2D cultures as measured by the amount of 63 hydroxylated product formed - a measure of Technology Massachusetts 'assachusetts Institute Institute of ofechnology -161 - - 161- Biotechnology Process Engineering Center Biotechnology Process Engineering Center activity of CYP's that are induced by PCN. The concentration of CLO and 3MC to be added to both the 2D as well as the 3D cultures were similarly optimized. CYP Induction Mediated by Nuclear Receptors P450 inducing agents Prototypic responsive rat liver CYPs Polycyclic aromatic hydrocarbons Phenobarbital Dexamethasone Fibrate drugs Cholesterol Bile acidsb IAI, 2B1, 3AI, 4A1, 7AI 7AI Thyroid hormone P450 reductase A2, 1131 2B2 3A2, 3A23 4A2, 4A3 Receptor Ah receptor' CAR PXR PPARa LXRa FXR TR 'PAS transcription family member, not a nuclear receptor. b Inhibitors of CYP7A transcription. Figure 7-1: Prototypical inducers and nuclear receptors that mediate the induction of some of the important CYP450's found in rat liver (akenfrom reference[179]) The role of nuclear receptors in gene induction and regulation has been described in Figure 7-2. The binding of the heterodimer leads to the activation of transcription of the target gene with the xenobiotic response element. In essence, the mechanism of induction of CYP450's in the presence of xenobiotics may be considered to be composed of three main components - a cue (the xenobiotic), a signal (the cascadeof events thatfollow the initial binding of the xenobiotic to the nuclear receptor),and a response (the downstreamupregulationof the targetgene). Massachusetts Massachusetts Institute Institute of of Technology echnology -162- -162- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Xenochemlcal XenoR-RXR - Retinoids I4> CYP Trnscriptof 1--- ERx Figure 7-2: Role of nuclear receptors in CYP gene induction. Shown is the structure of a xenobiotic receptor-RXR heterodimer (e.g., CAR-RXR, PXRRXR, or PPAR-RXR) bound to two copies of a hexameric DNA response element based on the sequence AGGTCA spaced by Xnucleotides. The hexameric repeat can be arranged as a DR, ER, or IR motif, as indicated. Naturally occurring DNA response elements for these nuclear receptors are generally imperfect repeats of the AGGTCA sequence. The extent to which retinoids that bind to RXR synergize with the xenobiotic in stimulating CYP gene transcription is uncertain [179] 14 la I-O co $_ T 0 Basal IM PCN 10 nM PCN 100 nM nM PCN 100 pm PCN 10 pm PCN 100 pm PCN Figure 7-3: Dose response in 2D collagen sandwich cultures: Case of PCN Massachusetts Institute Massachusetts Institute of of Technology Technoogy - 163 - - 163- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 7.2 Inducibility of 2D and 3D cultures using 3MC and clofibric acid Figures 7-4 gives a schematic of the mechanism of induction of 3MC responsive genes. No significant difference is seen in the basal or induced expression of CYP1A2 between 2DCSW and 3DB cultures in response to 72 hr. incubation with a 5 FtM solution of 3-Methyl Cholanthrene (Figure 7-6 (a)). Again, even though it may not be prudent to compare fold induction data across different labs that use different media formulations, a survey of the literature was done to get an estimate of the range of fold induction data seen in different labs. F Induction of CYP450 1A2 72 hr. induction by 5 uM, 3-Methyl Cholanthrene, Culture Medium replaced everyday. / / 9 Metaolism f nd v CYP1 rms ~~~~~~Mtaboisam of Iinand by CYPi fnrmns -- ".- - --1 - -- -· (ED "1QII'hMA1 , %.a0 -affIma- N a k -R t 3-Methyl Cholanthrene ARNT 4 AHR ligand _ AHR CUE L \I- -~ Increased CYPlA1, CYP1A2, transcription CYPiB 1 A I / j ?, \r - RESPONSE Figure 7-4: Mechanism of induction of 3MC-responsive genes The fold induction seen in 2DCSW and 3DB cultures of CYP1A2 at the mRNA level (9-14 fold over basal) is comparable to that reported in some in vivo studies in SpragueDawley rats, at both the activity and mRNA levels [143, 152]. Technology Massachusetts Institute Institute of ofTechnology 164- 164- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Figures 7-5 gives a schematic of the mechanism of induction of Clofibric Acid responsive genes. Fold induction relative to basal as well as the induced expression relative to in vivo of CYP4A1, in response to a 72 hr. incubation of a 100 1LMconcentration of Clofibric acid, are not significantly different between 2DCSW and 3DB cultures (Figure 7-6(b)). Induction of CYP450 4A1. 4a3 by Clofibric Acid 72 hr. induction by 100 uM, CLO, Culture Medium replaced everyday. /r F ---- cyp4a "SIGNAL" 1* j (7j 41 PPARa ligand r-I Increased r1YP4al VP4AS(P4AS~~~~~~~~ transcription v CUE . - JA , j -11-11 RESPONSE I Figure 7-5: Mechanism of induction of Clofibric Acid-responsive genes The fold induction seen in 2DCSW and 3DB cultures of CYP4A1 at the mRNA level (25-30 fold over basal) is comparable to that reported in some in vivo studies in SpragueDawley rats, at both the activity and mRNA levels [143, 152]. However, the basal expression of CYP4A1 is significantly down regulated (nearly 3-fold on a log2 scale, p<0.05) relative to in vivo,in 2DCSW cultures. The basal expression of 4A1 in 3DB cultures is comparable to that seen in vivo (p>0.95). MassachusettsInstitute of echnology - 165- Biotechnology Process Engineering Center 1 A2 6 4 4A1 +/- 1.2 , (b) (a) 5.9 +1- 0.8 4 2 0 ; ., ' | -2 -4 3.7 +/- 0.2 3.1 +/- 0.6 ·4 . .. i CO 72 hr. induction by 5 uM 3MC, Medium changed everyday, N=4, n=3 U - .. o 0 72 hr. induction by 100 uM CLO, Medium changed everyday, N=4, n=3 -6 Figure 7-6: Results of prototypical induction studies: 3MC, CLO studies (a): No statistical difference seen (p<0.0 5 ) in basal expression, induced expression relative to in vivo as well as in fold induction over basal of CYP1A2, CYP4A1 between 2D and 3D cultures. Data from N=4 different rats, with at least 3 replicates of each culture in each experiment (b): No statistical difference seen (p<0.0 5 ) in induced expression levels relative to in vivo as well as in fold induction over basal expression, between 2DCSW and 3DB cultures. Basal expression of CYP4A1 is seen to be significantly downregulated (2.9 fold on a log2 scale) in 2D over in vivo and 3D cultures (p=0.01). Data from N=2 different rats, with at least 3 replicates of each culture in each experiment 7.3 Inducibility of 2D and 3D cultures using PCN Induction of CYP450 3A is used as an important yardstick to judge new chemical entities (NCE's) during pre-clinical testing. The rat CYP3A1 and CYP3A2 enzymes share many properties of human CYP3A4, a prominent and inducible pathway for human drug metabolism. Figure 7-7 gives a schematic of the nuclear receptors and downstream signalling cascade events that lead to the upregulation of the PCN responsive genes. ecnolgyMasacusetsInsitte Miassachusetts Institute ofTechnology f 16 -166- Bitehnoog Poces ngieeingCete Biotechnology Process Engineering Center Induction of CYP450 3A1 by 16-Preanenolone Carbonitrile 72 hr. induction by 10 uM, PCN, Culture Medium replaced everyday. /101", ~~MPtahnlismnf inand hv CYP3A fnrmc F "SIGNAL" PXR ligand CUE - -. 31 i11) ) Ix Increased CYP3A1,3A2 transcription OATP2, CYP2C6 L \1 RESPONSE Figure 7-7: Mechanism of induction of PCN responsive genes Massachusetts Institu-e ofTechnology - 167- Biotechnology Process Engineering Center 6 4 2 0 -2 -4 -6 -8 -10 -12 -14 Figure 7-8: Results from the PCN Induction studies Significant downregulation (p<0.05) in basal expression of CYP2C6, 3A1 and 3A2 is seen in 2DCSW cultures relative to in vivo and 3D cultures. However basal expression of OATP2 is seen to be significantly downregulated (p<0.05 ) in 2D as well as 3D cultures, relative to in vivo. Both the induced expression relative to in vivo as well as the fold induction over basal of the genes CYP2C6, 3A1, 3A2 and OATP2 is seen to be significantly (p<0.05) higher in 3D cultures over 2D cultures. Data from N=3 different rats, with at least 4 replicates of each culture in each experiment Basal levels of both CYP3A1 and 3A2 are strongly downregulated in 2D culture (2-9fold) and are unchanged in 3D culture compared to in vivo (Chapter 6, Figure 6-13, & Figure 7-8). When induced with 16a Pregneleone Carbonitrile (PCN), the expression of 3A1 increases 7-fold in 2D culture, but remains strongly downregulated (2.6 fold). In contrast, CYP3A1 expression in 3D culture shows basal levels comparable to in vivo and is induced 39fold (or 5.3-fold on a log2 scale - Figure 7-8) above basal - comparable to the levels reported for in viv induction [160]. CYP3A2, though showing comparable -5-7 fold induction over respective basal levels in 2D and 3D, remains strongly downregulated in 2D (induced level is <5% of in vi). Thus, while enzyme induction is observed in 2D culture for this important enzyme family, the absolute levels are far below physiological, and thus any downstream consequences of metabolism (e.g., toxicity events) would not likely be reproduced in the 2D culture system. The 3D microreactor system provides an approximately physiological level of Massachusetts Institute Mlassachusetts Institute of of Technology Technology - 168168- Biotechnology Process Engineering Center Biotechnology Process Engineering Center induction for CYP3A2, perhaps through better maintenance of cytoskeletal integrity needed for optimal CYP3A induction [80]. Two other enzymes induced by PCN, CYP2C6 and OATP2, also show more physiological behavior in the 3D microreactor than in 2D. CYP2C6 is mod and CYP3Arately downregulated and is not inducible in 2D culture, but shows normal basal level of expression and 15-fold induction in 3D culture. OATP2 is strongly downregulated in both 2D and 3D culture, but is more highly inducible in 3D cultures (16fold) compared to 2D cultures (2.6-fold). 7.4 Key regulatory mechanisms that affect p450 basal expression and induction Other studies in literature have also shown that there may only be minimal differences in the response of a 2DCSW culture or a 3D static tissue system to the addition of the prototypical inducers of CYP1A and CYP4A genes [80]. Our data supports the hypothesis that there are negligible differences in the extent of induction seen in 2D and 3D cultures in response to prototypical inducers of CYPIA and CYP4A.. Interestingly, unlike previous studies we notice that the basal expression of CYP4A1 is higher in the 3D perfused bioreactor cultures (and closer to in vi levels) than the levels seen in the 2DCSW cultures. No such differences are seen in the basal expression level of CYP1A in 2D and 3D cultures. However, CYP1A is known to be pretty stably expressed in a number of different culture systems, and has even been seen to be not very sensitive to the culture conditions used [80]. Remarkably, the basal expression of CYP3A1, 3A2, and 2C6, are seen to be expressed at levels very close to that seen in the in vivo liver in the 3DB culture (p>0.95), while the basal expression levels of these genes are highly down regulated in 2DCSW cultures (p<0.05). This may be attributable to the better maintenance of key liver transcription factors in the 3DB (Chapter 6), at levels close to those seen in viw. Also, the induced expression relative to in vim, and therefore the fold change with respect to basal of CYP3A1, 3A2, and 2C6, is seen to be higher in the 3DB cultures than in the 2DCSW cultures. It has been shown in literature that cytoskeleton integrity is a pre-requisite for optimal CYP3A induction [183] and studies in literature have emphasized the need for matrigel substrata or a sandwich configuration for optimal induction of 2B and 3A genes [80]. Previous studies have concluded that while both 2D sandwich cultures as well as 3D cultures on Matrigel, allow increased longevity of rat hepatocyte cultures and optimal 169 of Technology Ins~~~tute Massachusetts - MassachusettsInstitute ofTechnology - 169- - Center~~~~ ProcessEngineering Biotechnology Biotechnology Process Enineering Center induction of CYP3A's and CYP2B's, constitutive CYP dependent activities were all drastically decreased in possibly all primary culture configurations [80]. However, unlike other 3D tissue systems used in literature, the 3D perfused microfabricated bioreactor cultures helps maintain even the basal CYP mRNA levels of a large number of very common CYP's at levels similar to those seen in vivo.In addition, we see a much higher induction of CYP3A's and CYP2C6, in the 3D bioreactor cultures, in response to PCN. The gene expression of nuclear receptors that mediate the induction of CYP's through prototypical inducers, in in ivo liver, isolated hepatocytes, 2DCSW cultures and 3DB cultures were also(Figure 7-9). AHR CAR PXR FXR RXRax PPARa 4 2 0 T LI ............................... ....f.....j.............T....... T" ................. IL4I .. 1._.'._._.. ...I -2 , I -4 -6 I 1 -R -n -103 Ml IbUIslaLU Hepatocytes I 2D collagen 2D collagen sandwich cultures 3D 3D3 microreactor cultures Figure 7-9: Basal expression of ligand-binding nuclear receptors Significant downregulation (p<0.05) in expression of CAR, PXR and FXR seen in the 2D collagen sandwich cultures. HNF4a has been shown to regulate expression of CAR and PXR [184]. Downregulation of CAR may be one reason for the differences seen in the inducibility of the 2D collagen sandwich cultures and 3D microreactor cultures by PCN. Data from N=3 different rats, with at least 4 replicates of each culture in each experiment. Interestingly, the expression of AHR, RXRa and PPARa that mediate the induction of CYP1A2 and CYP4A1 are comparable in both 2DCSW and 3DB cultures and similar to expression levels seen in vivo. However, the expression of PXR, that has been shown to mediate the induction and regulate the basal expression of CYP3A1, 3A2, 2C6 and OATP2 Massachusetts Massachusetts Institute Institute of of Technology echnology 170 - 170- Biotechnology Process Engineering Center BitechnologyProcessEngineringCenter [179, 184, 185] is seen to be downregulated in 2DCSW cultures compared to in vivo and 3D bioreactor cultures. HNF4a has been reported in literature to regulate the expression of CAR and PXR [184]. Thus, we may hypothesize that the differential levels of induction seen in 2DCSW and 3DB cultures even at high non-toxic concentrations of PCN that give maximal induction in 2D cultures, may be due to differences in the expression of the liver transcription factor HNF4 between 2D and 3D cultures which regulates the expression of the nuclear receptors CAR and PXR that bind to the prototypical inducer. Thus the 3D microfabricated perfused bioreactor is more sensitive to the addition of prototypical inducers that cause the over-expression of CYP3A, 2C6 or OATP2's, than 2DCSW cultures. A large majority of NCE's and xenobiotics are metabolized by or induce the CYP3A class of enzymes - thus the bioreactor may serve to be a more sensitive model than the 2DCSW culture systems, as a screen for CYP3A metabolism or inducibility studies. In addition, unlike most static 3D culture systems (e.g. 3D matrigel spheroids) where the basal expression of a number of Phase I and Phase II genes have been reported to be significantly down regulated, independent of the nature of the extra cellular matrix environment used in the primary cultures [80]., the ability of the 3D perfused microfabricated bioreactor to maintain the basal expression of many of the Phase I and Phase II genes at levels very similar to those seen in vivo, indicates the suitability of the system as a tool for xenobiotic toxicity studies and also points to the important role that perfusion may play in maintaining liver-specific gene expression. 7.5 Relative inducibility of 2D and 3D cultures - a summary Induction of CYP450's is used as an important yardstick to judge new drugs during pre-clinical testing. The rat CYP3A1 and CYP3A2 enzymes share many properties of human CYP3A4, a prominent and inducible pathway for human drug metabolism. Basal levels of both CYP3A1/2 are strongly downregulated in 2D culture (2-9- fold) and are unchanged in 3D culture compared to in viW (Figure.6-12, Figure 7-8(a)). When induced with 160a pregneleone carbonitrile (PCN), the expression of 3A1 increases 7-fold in 2D culture, but remains strongly downregulated (2-6 fold). In contrast, CYP3A1 expression in 3D culture shows basal levels comparable to in tivoand is induced 39-fold (or 5.3-fold on a log2 scale in Figure 4a) above Massachusetts Massachusetts Institute Institute of of Technology echnology - 171 171- - Biotechnology Process Engineering Center Biotechnology Process Elgineering Center basal - comparable to the levels reported for in vivo induction [160]. CYP3A2, though showing comparable -5-7 fold induction over respective basal levels in 2D and 3D, remains strongly downregulated in 2D (induced level is <5% of in vim). Thus, while enzyme induction is observed in 2D culture for this important enzyme family, the absolute levels are far below physiological and thus any downstream consequences of metabolism (e.g., toxicity events) might fail to be reproduced in the 2D culture system. The 3D microreactor system provides an approximately physiological level of induction for CYP3A2, perhaps through better maintenance of cytoskeletal integrity needed for optimal CYP3A induction [80]. Two other enzymes induced by PCN, CYP2C6 and OATP2, also show more physiological behavior in the 3D microreactor than in 2D. CYP2C6 is moderately downregulated and is not inducible in 2D culture, but shows normal basal level of expression and 15-fold induction in 3D culture. OATP2 is strongly downregulated in both 2D and 3D culture, but is more highly inducible in 3D cultures (16-fold) compared to 2D cultures (3-fold). CYP1A2 is among the most robust and inducible of the CYP450's [80] and exhibits no significant difference in basal or induced expression levels in 2D compared to 3D culture, and the magnitude of induction (Figure 7-6(a), 9-14 fold over basal) is comparable to that reported in vivoin Sprague-Dawley rats, at both the activity and mRNA levels [143], [186]. These results are consistent with previous reports of the relative insensitivity of induction levels of the CYP1A and CYP4A genes in 2D collagen gel sandwich or 3D static culture [80]. The pattern of near-normal physiological CYP450 induction in 3D culture with subnormal behavior in 2D culture is observed for clofibrate induction of CYP4A1 (Figure 76(b)). Although the magnitude of induction above basal levels appears greater for 2D culture (60-fold) than for 3D culture (25-fold), the initial basal level in 2D culture is moderately downregulated in 2D culture but unchanged compared to in vivo for 3D microreactor culture, and thus the induced level in 2D is far below the induced level in 3D. The magnitude of induction in 3D is comparable to the value of 25-30 fold over basal observed in Sprague-Dawley rats in viv at both the activity and mRNA levels [143, 152]. Previous studies have concluded that while both 2D sandwich cultures as well as 3D cultures on matrigel allow increased longevity of rat hepatocyte cultures and optimal induction Massachusetts Institute ofTechnology -172- Biotechnology Process Engineering Center of CYP3A's and CYP2B's, constitutive CYP450 dependent activities were almost all strongly decreased in possibly all primary culture configurations [80]. The results for 2D collagen gel sandwich cultures in the induction experiments done as part of this thesis are consistent with that conclusion. However, the same experiments also show that near-physiological basal and induced levels of most CYP450s can be achieved in 3D microreactor culture. This result is consistent with the more physiological expression levels of key liver-enriched transcription factors and suggests that perhaps broader programs of liver-specific gene expression may be obtained in 3D microreactor culture (Figure 6-13). Based on the induction results, it is expected that the expression of the nuclear receptor PXR that mediates the basal and induced expression of PCN-inducible enzymes [179, 184, 185] be more physiological in 3D than in 2D culture. Indeed, this is the case (Figure 7-9). These results are also consistent with better retention in 3D of the liver-enriched transcription factor HNF4a, which regulates the expression of CAR and PXR [184]. Expression levels of nuclear receptors that mediate the induction of CYP1A2 and CYP4A1 -- AHR, RXRa and PPARa -- are comparable in both 2D and 3D cultures and similar to expression levels in vivo,a result consistent with the expression patterns of CYP1A2 and CYP 4A1. The Farnesoid X receptor or FXR, that is known to regulate cholesterol synthesis and bile acid metabolism, is also maintained at near in vivolevels in the 3D microreactors unlike the 2D cultures where they are almost 4-fold downregulated on a log2 scale. 7.6 Summary of key results and conclusions The key results and conclusions that may be drawn from the set of inducibility studies that compared the basal and induced expression of CYP450's are: 1. Statistically, there is no difference in the basal and induced expression of CYP1A2 between the 2D collagen sandwich and 3D bioreactor cultures. The levels of induced expression of CYP4A1 relative to in tvi, are comparable in 2D and 3D cultures, however the basal expression of CYP4A1 is better maintained in 3D cultures than in 2D collagen sandwich cultures. Technology Massachusetts Massachusetts Institute Institute of ofTechnology - 173 - -173- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 2. The 3D bioreactors have been shown to be a more inducible (and therefore a more sensitive) system than the standard 2D collagen sandwich cultures, in response to the addition of the inducer Pregneolone 16a Carbonitrile. 3. The basal expression of key nuclear receptors, that regulate the basal and induced expression of CYP's are maintained are close to in viva levels in the 3D bioreactors. 4. Liver enriched transcription factors - specifically HNFla and HNF4 a - that regulate the expression of the ligand-binding nuclear receptors CAR and PXR, are maintained at close to in vim levels in the 3D bioreactor, but are downregulated in 2D collagen sandwich cultures. 5. The basal expression of the organic anion transporter protein - OATP2 is downregulated in both 2D and 3D cultures. This may be attributable to the absence of specific biochemical stimuli in the media solution used in cultures [21]. However, the 3D bioreactors still retain their ability to induce the transcription of OATP2 in response to the addition of the prototypical inducer PCN. 6. The 3D bioreactor may serve as a more sensitive in vitroscreen than standard 2D cultures to evaluate the induction potential of NCE's. Inttt fTcnlg Mascuet Massachusetts Institute ofTechnology 14 - 174- itchooyPoesEgneigCne Biotechnology Process Engineering Center Chapter 8 Conclusions and Recommendations This thesis focused on the design, fabrication, modeling and characterization of a microfabricated bioreactor system that mimics the key facets of the in tvio microenvironment in the hepatic acinus by allowing for the three dimensional morphogenesis of liver tissue under continuous perfusion conditions. A key feature of the bioreactor is the distribution of cells into many tiny (-0.001 cm3 ) tissue units that are uniformly perfused with culture medium. The total mass of tissue in the system is readily adjusted for applications requiring only a few thousand cells to those requiring over a million cells by keeping the microenvironment the same and scaling the total number of tissue units in the reactor. A computational fluid mechanical model in ADINA® was used to describe the velocity and shear stress distribution within the bioreactor. The model helped identify a set of crossflow (perfusion) flow rates that simulated physiological shear stress conditions at the tissue surface within a channel of the bioreactor. A mass transfer model in FEMLAB® was used to describe the tissue distribution of metabolite concentration gradients. The mass transfer model helped identify appropriate bioreactor operating conditions (Peclet and Damkohler numbers) under which the tissue oxygen demand was theoretically satisfied. These predicted conditions were then experimentally validated, by looking at the expression of hypoxia-responsive genes as a function of perfusion rate. The mass transfer model may also be used to determine the tissue disposition of any metabolite, so long as the requisite model parameters - the Peclet Number and the Thiele Modulus or the Damkohler number are known. It may also be used to determine the intrinsicreactionrate given the experimentally measured apparent reaction rate at non-saturable concentrations of the drug (that has embedded effects of mass transfer) or given the steady state concentration of the product of reaction at the reactor outlet. (measured in a one-pass through configuration of the reactor). However, the MilliF reactor with - 20,000-40,000 cells would not able to produce enough (or detectable) amount of product metabolite in a one pass operation of the reactor given the Technology Mas5achusetts assachusetts Institute Institute of ofTechnology 175 -175- - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center reaction rates of most enzyme-substrate reactions of interest to the Pharmaceutical industry. The giant reactor which accommodates at least 250 times the cell numbers seeded into the MilliF reactor, may be used in a one-pass mode to get first pass apparent extraction rates that can then be used in the mass transfer model to determine the intrinsicreactionrate.However, the giant reactor must be operated in a one-pass back mix mode, so that the reactor volume (now equal to the volume of the reactor plus the recycle loop) operates like a CSTR, similar to the fluidic design suggested by Catapano and co-workers [187, 188]. The reactor volume (and hence the reactor residence time) must be minimized so that local changes in concentration are quickly equilibrated throughout the system. This fluidic design can help ensure that every channel in the reactor sees a constant concentration of the drug, even at low doses. A schematic of the fluidic design that may be used in the giant reactor is described below in Figure 8-1. One Pass Back mix Reactor Qin + R, C Qout = Qin, Cout ) 111 111t I I 11 'W" 2000 als / channel 20(06 clla I C I channel( -I Recycle Ratio Qr=- As Qr> > 1, Cout - C', at steady Qin Back mix rxtr.: as Qr state O0Rxtr. has PFR characteristics as Qr -oo Rxtr. has CSTR characteristics Figure 8-1: A one-pass back mix fluidic design for the measurement of one-pass extraction rates at non-saturable concentrations of the drug f echolgy MasacusetsInsitte Massachusetts Institute of Technology 17- - 176- Botchnloy Pocss ngneein Cete Biotechnology Process Engineering Center Using a broad spectrum of gene expression, protein expression and biochemical activity metrics, the liver tissue phenotype maintained during culture in the 3D bioreactor was seen to be substantially closer to that of native liver than that of cells maintained in standard in tite cultures, especially the 2D collagen gel sandwich cultures.. Global transcriptional profiling was first used to identify genes that are differentially expressed between 2D collagen sandwich cultures (2DCSW) and a variety of 3D cultures, including the 3D bioreactor (3DB), at multiple time points up to 20 days in culture, relative to the expression profiles seen in liver in tvio and compared to primary isolated cells. From these studies, a specific time point to compare the basal and drug-induced expression levels of several important CYP450 and Phase II metabolism genes more quantitatively via RT PCR and biochemical activity assays was identified. Semi-quantitative RT PCR assays were used to compare basal expression levels of key hepatic nuclear receptors and transcription factors that regulate the basal and induced expression of the CYP450 genes and other broad programs of liver-specific genes. It is hypothesized that the better preservation of the basal expression of the hepatic transcription factors may possibly be responsible for the more in vivo like expression of the liver specific genes in the 3D microreactor than in the 2D collagen sandwich cultures. In addition, the 3DB cultures were also found to be more sensitive to induction by certain specific prototypical inducers than the standard 2D collagen sandwich cultures. The better preservation of the ligand-binding hepatic nuclear receptors in the 3D bioreactor is one possible reason for the more sensitive response of the 3D bioreactor in response to the addition of inducers, than the 2D collagen sandwich cultures. Genes that are likely regulated by soluble factors present in the media were downregulated in both the 2D collagen sandwich as well as the 3D microreactor cultures. Such genes include the CYP2E1 (regulated by insulin in the media), and specific transporters - the Bile Salt Export Pump (BSEP), the organix anion transporter (OATP1 and OATP2), and the multi drug resistance gene (MDR2). It is hypothesized that the addition of specific biochemical stimuli such as HGF (Hepatocyte Growth Factor) may help preserve the expression of the specific transporter genes that are present specificallyin the bile duct cells. In addition, data from Dr.Kevin Leach, a Research Scientist in the Tannenbaum Lab, have shown improved Massachusetts assachusetts Institute Institute of of Technology Technology 177- 177- Biotechnology Process Engineering Center Biotechnology Process Engineering Center maintenance of the hepatic transcription factors and the CYP genes as a result of the addition of anti-oxidants to the 2D collagen sandwich cell culture (either immediately after cell isolation form the rat liver or at the time of plating). Antioxidants are known to help scavenge reactive oxygen intermediates that are generated due to ischemic-reperfusion injury following liver perfusion and cell isolation [80, 145] and can thus help minimize the downregulation of the CYP proteins that is primarily caused due to the nitric oxide and/ or peroxynitrites produced during isolation that causes specific blockage of the hemes of the CYP450s, resulting in alteration of enzyme activities in vitro through mechanisms downstream of mRNA [151]. A possible future work would be to study the effect of the addition of antioxidant treated cells (or spheroids) into the bioreactor. It is hypothesized that this should help improve the functional phenotype of the tissue in the bioreactor, as well as the tissue phenotype of the spheroids formed from these cells. This study has attempted to show the utility of three dimensional tissue constructs as a more predictive screen for metabolism and toxicity, by looking at a very broad set of fundamental tissue phenotypic as well as regulatory markers. The study has also identified key regulatory mechanisms that may be responsible for better retention of hepatic xenobiotic biotransformation phenotype in 3DB cultures over 2DCSW culture. We hypothesize that the 3D liver tissue constructs - because of their more in ivo like phenotype seen in short-term as well as in long-term cultures; could also serve to fulfill the unmet need of suitable in vitro models to study disease progression such as Hepatitis C. In addition, the microfabricated 3D bioreactors are poised to be used to test and quantify the toxicity of known pharmaceutical liver toxins such as Troglidazone, Acetaminophen, etc. The capacity of the 3D liver tissue bioreactor system to maintain tissue phenotype over long periods in culture (at least upto 3 weeks) posits a hypothesis that they may be more responsive to chronic toxins, and those not amenable to study by cryopreserved hepatocytes. Clinically, chronic overdose outcome is inferior to intentional overdose, suggesting an important role for assessment of chronic toxicity. In addition, a key set of experiments would be to test for the toxicity of compounds (sourced from Pharmaceutical companies) in the rat 3D bioreactor model, of compounds that were found to be toxic in human clinical trials, but not in traditional in vitm screens used by the fTcnlg Inltt Massachusetts Massachusets Institute of Technology 7- 178 Prcs EnierngCne Bitcnlg Biotechnology Process Engineering Center Pharmaceutical industry. This can be done pending the ready availability of human cells for use in the bioreactor. The use of human cells in the bioreactor would help open up new application areas for the bioreactor. While the biophysical and biochemical conditions that may be needed to help maintain the tissue phenotype of primary human cells may be different from those needed for primary rat hepatocytes, some of the learning derived from the use of rat hepatocytes in the system can help accelerate the process of optimization of biophysical and biochemical conditions for the retention of healthy human tissue phenotype in the bioreactor. The human tissue constructs created in the 3D bioreactor may be used to test for the toxicity of known human liver toxins. Concurrently, xenobiotics that were found to be toxic during late stage human clinical trials, may be tested on the human liver tissue bioreactor to determine the ability of the system to pick up the toxicity of such compounds. The in tivo like microenvironment provided by the bioreactor also opens up the possibility of its use as a tool to detect and isolate biomarkers. Genomics, Proteomics and Metabolomics studies on a disease-modeled bioreactor tissue or a toxin-responding bioreactor tissue [189] can help provide a 'systems-biology' approach to the identification of biomarkers [190-193]. However, the industrial use of the bioreactor for one or more of the above high throughput applications will require its re-design into a 6/24/96 well plate 3D perfused tissue format. Such a format, will allow the system to transition from a 'high information content' assay system to a 'high throughput, high information content' assay system. Massachusetts Institute ofTechnology -179- Biotechnology Process Engineering Center References 1. newestimatesof DiMasi, J.A., R.W. Hansen, and H.G. Grabowski, Theprice of innovation: drug developmentcosts.J Health Econ, 2003. 22(2): p. 151-85. 2. Annu Rev Pharmacol Davila, J.C., et al., Predictivevalueof in vitromodelsystemsin toxicology. Toxicol, 1998. 38: p. 63-96. 3. 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Moncion, A., et al., Identificationof a 16-nucleotidesequencethat mediatespost-transcriptional regulation of rat CYP2E1 byinsulin.J Biol Chem, 2002. 277(48): p. 45904-10. 145. Tirmenstein, M.A., et al., Characterigationof nitric oxideproductionfollowingisolation of rat hepatoytes. Toxicol Sci, 2000. 53(1): p. 56-62. 146. Kocarek, T.A., E.G. Schuetz, and P.S. Guzelian, Expressionofmultipleformsofrtochrome P450 mRNAs in primay culturesof rat hepatoytesmaintainedonmatrigel.Mol Pharmacol, 1993. 43(3): p. 328-34. 147. Allen, J.W. and S.N. Bhatia, Formation of steady-stateoxygengradientsin vitro:applicationto liver Zonation. Biotechnol Bioeng, 2003. 82(3): p. 253-62. 148. Liu, L., et al., Suffotransferasegeneexpressioninprimay culturesof rat hepato'ytes.Biochem Pharmacol, 1996. 52(10): p. 1621-30. 149. Yamagata, K., et al., Mutationsin the hepatocyte nudearfactor4alphagene in maturiy-onset diabetesof theyoung(MODY1). Nature, 1996. 384(6608): p. 458-60. 150. 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Meredith, C., et al., Studies on the inductionof rat hepatic CYP1A, CYP2B, CYP3A and rat liverslices. CYP4A subfamiyformmRNAs in vivoand in vitr usingprecision-cut Xenobiotica, 2003. 33(5): p. 511-27. 161. and in vitroand in vivo Handschin, C., M. Podvinec, and U.A. Meyer, In silicoapproaches, experimentstopredictinductionof drugmetabolism.Drug News Perspect, 2003. 16(7): p. 42334. 162. Ortiz de Montellano, P.R., Cyotchromep450: Structure,Mechanism and Biochemistry,ed. M.e. al. 1995: Plenum Press. 163. Park, B.K. and A.M. Breckenridge, Clinicalimplicationsof engymeinductionand enZyme inhibition. Clin Pharmacokinet, 164. 1981. 6(1): p. 1-24. Thomas, S.H., Paracetamol(acetaminophen)poisoning.Pharmacol Ther, 1993. 60(1): p. 91120. 165. Wei, P., et al., The nuclearreceptorCAR mediates specificxenobioticinduction of drug metabolism. Nature, 2000. 407(6806): p. 920-3. -192Institute of Technology Massachusetts Massachusetts Institute of Technology - 192- BiotechnologyProcess EngineeringCenter~ Biotechnology Process Engineering Center 166. Xie, W., et al., Reciprocalactivationofxenobiotic responsegenesby nuclear receptorsSXR/PXR and CAR. Genes Dev, 2000. 14(23): p. 3014-23. 167. Honkakoski, P. and M. Negishi, Regulationof ytochromeP450 (CYP) genesby nuclear receptors.BiochemJ, 168. 2000. 347(Pt 2): p. 321-37. Mangelsdorf, D.J., et al., The nuclearreceptorsupefamiy: the seconddecade.Cell, 1995. 83(6): p. 835-9. 169. Gonzalez, F.J. and Y.H. Lee, Constitutiveexpressionof hepatic ytochrome P450genes. Faseb J, 1996. 10(10): p. 1112-7. 170. MacDonald, C.J., H.P. Ciolino, and G.C. Yeh, Dibenoylmethanemodulatesayl hydrocarbon receptorfinction and e.pressionof ytochromes P50 1A1, 1A2, and B1. Cancer Res, 2001. 61(10): p. 3919-24. 171. Mangelsdorf, D.J. and R.M. 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Nippon Rinsho, 2000. 58(12): p. 2452-7. 179. Waxman, D.J., P450 gene induction by structurall diversexenocemicals: centralrole of nuckar receptors CAR, PXR, and PPAR Arch Biochem Biophys, 1999. 369(1): p. 11-23. Technology Massachusetts Massachusetts Institutc Institute of ofTechnology 193-193- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 180. Lin, J.H. and A.Y. Lu, Inhibition and induction of ytochromeP450 and the clinicalimplications. Clin Pharmacokinet, 1998. 35(5): p. 361-90. 181. Pelkonen, O., et al., Inhibition and inductionof human ytochrome P450 (CYP) enuymes. Xenobiotica, 1998. 28(12): p. 1203-53. 182. P450 by xenobiotics. Bresnick, E., R. Foldes, and R.N. Hines, Inductionoftytochrome Pharmacol Rev, 1984. 36(2 Suppl): p. 43S-51S. 183. ofapretranscriptional Brown, S.E., L.C. Quattrochi, and P.S. Guzelian, Characterization of ytochromeP450 3A23 inprimay culturesof adult rat pathwayforinductionbyphenobarbital hepatoytes. 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Technology Massachusetts MassachusettsInstitute Institute of ofTechnology 195 -195- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendices: Protocols and Experimental Methods Appendix 1: Isolation of primary rat hepatocytes using Percoll® Appendix 2: Isolation of total RNA from isolated hepatocytes, in vivotissue, 2D and 3D cultures Appendix 3: Primer design procedures and guidelines Appendix 4: cDNA Preparation and Reverse Transcriptase-Polymerase Chain Reaction (RTPCR) protocols Appendix 5: Hepatocyte isolation and Spheroid Formation Appendix 6: Measurement of the total number of viable cells in cultures using the measured amount of total RNA and RT-PCR against the 18s gene Appendix 7: Preparation of 2D collagen gel sandwich cultures Appendix 8: Preparation of 3D spheroid and 3D Matrigel spheroid cultures Appendix 9: Induction experiments on 2D collagen sandwich and 3D microreactor cultures Appendix 10: Accession numbers for mRNA and complete cds sequences used Appendix 11: Primer sequences used in RT-PCR studies Appendix 12: Melting and annealing temperatures of designed primers Appendix 14: Microarray Processing Appendix 15: Testosterone Metabolism studies Appendix 16: The Drug Development Process - an Overview fTcnlg Inttt Masacustt Massachusetts Institute ofTechnology 9 -196- itehooyPoesEgneigCne Biotechnology Process Engineering Center Appendix 1 Isolation of primary rat hepatocyte enriched fraction using Percoll Materials Needed: PercollU Solution (Sigma, P1644-1L), 10X HBSS (Gibco #14025-076), Trypan Blue (Gibco #15250-0), aluminum foil, haemacytometer 1) Add 21.6mL of Percoll to 2.4mL of 10X Hank's Balanced Salt Solution in a 50mL conical tube to create a Percoll concentration of 90% v/v and put on ice. (his must be done the day of the isolation because the salt in the HBSS begins to dissolve the Percoll silica beads when stored) 2) Dilute hepatocytes to a density of 5-7 million cells/mL in 24 mL of HGM. (Need a minimum 125 million hepatocytes). Higher cell densities decrease the efficiency of separation as contaminant cells are carried through the gradient and lower densities reduce the yield. 3) Gently mix 25 mL of cell suspension with 24 mL of the 90% Percoll suspension to form a uniform isotonic gradient. 4) Centrifuge at 50 x g for 10 min at 4"C in a swinging bucket centrifuge (maximum acceleration and brake setting of 3) 5) Aspirate the dead and compromised cell from suspension, and re-suspend the resulting pellet in 45 mL of HGM. 6) Centrifuge at 50 x g for 3 min at 4"C in a swinging bucket centrifuge. Remove the supernatant and re-suspend cells in the appropriate volume of HGM. Determine cell count and viability using trypan blue exclusion on a hemacytometer. Keep cells on ice until use, but try to process hepatocytes immediately, as they begin to dedifferentiate rapidly in suspension Technology Massachusetts Massachusetts Institute Institutet of ofTechnology 197--197 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendix 2 Isolation of total RNA from isolated hepatocytes, in vivo tissue, 2D and 3D cultures Materials Needed: Trizol Reagent (Gibco #15596-026), Chloroform, Qiagen RNeasy Mini Kit (Qiagen #74104), 100% Ethanol, Cell scraper, Electric blade homogenizer, RNase-free water (such as DEPC-treated water), and RNase-free tips and tubes, ice bucket with lots of ice, 25gage needle, 3 mL syringe Day 1: Collection of in vivo tissue samples, isolated hepatocytes samples, 2D and 3D samples For isolated hepatocytes 1) Re-suspend final known number of viable isolated hepatocytes in HGM at a density of -1 million cells/mL and place in a 1.5 mL eppendorf tube. 2) Centrifuge at 50g for 3 min. at room temperature 3) Aspirate medium using a 1000 tL pipetteman set to 800 iL being very careful not to touch or suction the media-cell interface. 4) Add 1 mL of Trizol® reagent to the eppendorf tube 5) Mix the cells thoroughly with TRIzol®, use a 25guage needle to aspirate and pipette the TRIzol® 15-20 times within the eppendorf tube. This helps homogenizing the cells. 6) *** store sample for further processing in the -80"C for upto 1 month **** (note - Qiagen sells RNALater® a preservation media solution that needs to be used in case the RNA samples will be stored for more than a month) For in vivo tissue samples 1) Surgically remove 1-3 cm thick tissue samples (slices) from the rat liver taking care to remove multiple samples from different lobes of the liver. Technology Massachusetts Massachusetts Institute Institute of ofTechnology 198 -198- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 2) Transfer the samples into separate centrifuge tubes (one tube for each lobe) filled with TRIzol® (use - 2 mL of TRIzol® for each 1-3 cm thick piece) 3) Using a rotary electric blade homogenizer (in the Tannenbaum Lab) completely homogenize the in vivo tissue samples in the four centrifuge tubes. The solid tissue samples should be homogenized till very few solid pieces are seen in the centrifuge tubes. The homogenization needs to be done thoroughly, else some of the RBC's (red blood cells) that are left unhomogenized can clump together and clot. 5) *** store sample for further processing in the -80"C for upto 1 month **** (note - Qiagen sells RNALater® a preservation media solution that needs to be used in case the RNA samples will be stored for more than a month) For 3D spheroids 1) Size separate the spheroids of the desired range (usually 100-300 glm),centrifuge the spheroid solution at 50g for 3 min., aspirate the media out and add 1 mL of TRIzol® per million cells taken from the spinner flask (i.e. roughly for every 3 mL of a 30 million cells seeded spinner flask). Using a 25gage needed attached to a 3 mL syringe, homogenize the spheroid sample till a clear solution is seen. 2) *** store sample for further processing in the -80"C for upto 1 month **** (note - Qiagen sells RNALater® a preservation media solution that needs to be used in case the RNA samples will be stored for more than a month) For 2D collagen sandwich cultures and 3D Matrigel® cultures 1) Aspirate the medium out of the wells 2) Add 5001I of TRIzol® solution to each well of a 6 well plate. The volume can be scaled accordingly for tissue culture plates of other sizes. 3) *** store sample for further processing in the -80"C for upto 1 month **** (note - Qiagen sells RNALater® a preservation media solution that needs to be used in case the RNA samples will be stored for more than a month) Technology Massachusetts Massachusetts Institute Institute of ofTechnology - 199 - - 199- Biotechnology Process Engineering Center Biotechnology Process Engineering Center For 3D cultures 1) Disassemble the reactor and transfer the cell-scaffold into an eppendorf filled with 500-750 Vl of TRIzol® (in general 1 mL of TRIzol® is added per million cells, but adding too much of TRIzol ® is not a big issue) 2) Using a 25gage syringe attached to a 3 mL syringe, aspirate and pipette the TRIzol® through the cell-scaffold at least 15-20 times, to dislodge the cells from the scaffold 3) Take a look at the silicon chip under the microscope to make sure that the cells have indeed been removed from the scaffold. In the rare cases when some cells still remain, place the eppendorf with the scaffold in the sonicator and sonicate the sample for 5 minutes to remove all cells from the scaffold 4) *** store sample for further processing in the -80"C for upto 1 month **** (note - Qiagen sells RNALaterx a preservation media solution that needs to be used in case the RNA samples will be stored for more than a month) Day 2: Isolation of RNA from the in vivo, isolated hepatocytes, 2D collagen sandwich, 3D Matrigel® and bioreactor samples For in vivo samples - take not more than 500 L of the homogenized sample stored at -80"C (samples take 15 min. to thaw), from each centrifuge tube and transfer into a 1 mL eppendorf tube For the case of 2D collagen sandwich cultures - thaw the plates from the -80°C freezer, use a cell scraper to scrape the collagen and cells off the plate. Use a 25 gage needle attached to a 3 mL syringe, to homogenize the collagen-cell solid pieces that remain after scraping - the homogenization must be done thoroughly - no portion of the collagen-cell solid pieces must be visible after homogenization. It takes nearly 45 min. to thoroughly homogenize one 6-well plate. The homogenized solution from each well must be transferred to a separate 1 mL eppendorf tube. For the 3D Matrigelf cultures: repeat all the steps for the 2D collagen sandwich cultures All other samples that are already in eppendorf tubes (isolated hepatocytes samples and 3D bioreactor samples) can be removed from the -80"C freezer and thawed. Technology Massachusetts Institute Massachusetts Institute of ofTechnology - 200 200- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center 1) Add 200 1iL (add 250 gLl/ mL of TRIzol® per mL of TRIzol® though there is no disadvantage of adding more - only that the downstream steps will take longer as more volume of sample needs to be treated for RNA extraction and cleaning) of Chloroform to each eppendorf tube. 2) Vortex for 30 sec (Solution should become cloudy pink and be consistent throughout). It will slowly phase separate. Leave solutions to stand at for 2 min. at room temperature. 3) Centrifuge in the cold room (4"C) at 12,000 rpm for 15 min to allow the samples to separate into 3 phases. Transfer the samples onto ice. 4) Take upper clear, aqueous phase into a new 1.5 mL eppendorf tube paying particular attention not to aspirate any of the pink interphase, using a 200 L pipetteman (approx. acquire 400-700 FL of clear aqueous solution of RNA is recovered per mL of TRIzol ) (NOTE - The bottom organic phase contains protein and DNA that will reduce the quality/purity of you RNA sample if aspirated along with the RNA). Make sure that the samples are on ice. Replace the un-used protein and DNA fractions into a cryobox and place the same back into the -80"C freezer 5) Add an equal volume of 70% EtOH to the aqueous phase and pipette the solution up and down till a clear solution emerges. 6) Label RNEasy® filter columns provided in the RNEasy® Mini Kit - one for each sample, add 650 yIL (capacity of the columns is 700 VtL)of the ethanol-RNA solution, and centrifuge for 15 sec. at 2000 rpm (room temperature). 7) Capture the column elutant in the collection tube and pass the collected elutant through the same filter column once again i.e. repeat step 12, then discard elutant. 8) Repeat steps 12 & 13 until all RNA solution has been passed twice through the filter column 9) Discard collection tube, replace the RNA containing filter column into .a clean new RNasefree collection tube (provided in the kit) 10) Add 500 jIL of RPE Buffer (provided in the kit, add ethanol to the RPE stock solution as indicated on the bottle top - this has to be done when a new RPE bottle is used - ethanol has Technology Massachusetts MassachusettsInstitute Institute of ofTechnology -201-201 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center to be added only once to the RPE stock, RPE stock diluted in ethanol can be stored at room temperature) to each filter column and spin for 15 sec (this helps dean the RNA out removes the contaminating DNA present in the sample) Qiagen manual, pg 80 - Replacing the collection tube before the RPE wash is essential because the RNA in EtOH is 'dirty' from the isolation process. The RPE buffer is cleaning the RNA, and if same collection tube is used, the cleaning protocol can be compromised 11) Add 500 jiL more of RPE buffer, spin for 1 min, and discard the elutant. Centrifuge the empty filter column with bound RNA, with a collection tube at the bottom, for 1 min. This can help remove the last bit of RPE that remains in the filter. 12) Place column in a fresh 1.5 mL eppendorf (provided in the kit), add 30 L of RT-PCR grade water (or DEPC water or Tris water) to the filter, taking care that the entire filter area is wetted, and spin for -30 sec 10,000 rpm 20) The collected RNA solution may be checked for purity and quality, by measuring the A260/ A280 reading in a spectrophotometer. Store RNA solution at -80"C - for long term (more than a month's storage, it is advisable that the RNA is stored in a RNALater® stabilizing agent). ~ Massachusetts~~ Massachusetts Institute of Technology ~~ ofTcnlg 22-BoehooyPoesEgneigCne Inttt Biotechnology Process Eineering -202- Center Appendix 3 Primer design procedures and guidelines Step 1: Identify Gene Sequence > Use NCBI Nucleotide Search (http://www.ncbi.nlm.nih.gov/) to find the target genes' mRNA sequence, as far as possible use complete cds or the mRNA sequence. Indude species (rattus nonvegicus)and RNA type (i.e. mRNA, rRNA, etc.) in your search >' Step 2: Dengn Initial Pnimer > Input the resulting gene sequence into MITs Primer3 software (Whitehead Institute) Specify the following conditions (shown in the field below as well) o 100-200 basepair target sequence o o 18-30 nudeotides in length 45-55% GC content C·n·...nnn-..·allman.l··a. La Vc I ts Ba 4· Ftes lo tP ' .J L-1 d- - j-'' , - - I Ulrdes tto9bktmlppuvsd cdShSo.tsJ3 Got" - f %9wchd - % 8 rR0 - R '41.i' n GO*- posseS p-W3 V *Se~doWlb *4hSecid. SW W 4) .-- 9 .- 00 a .. Pq. . -- .- .. V n: , @a O .. n L,: LI 1oga-A _ I g~ ' ~i - 1' -aaI Ptb une mquear w(Y3,, .tzt oACO_-_.t._-otha==' ttotd 04idc bbu t FASTA tomtok Pua Not ,t.cJoru,_,...e nai L ._, ltoro! NONE 961 catCsgggcat caccogggtc ttcatcaat rgogatct ctctggeatc acagtggatg 1021cccocotgs gettoagcaggctgtgcata aggctutgct gaccttagat gagaIgggga W.: 'r1t.i , · ebLqn. (wcto.AWUe, LJNE 1081ctlqgstgc qgoagcctSctr g tggaggccgtcc t gttotgtcc cc ctc tgae cccctctttg 1141attctcgacca cccottret tcargag ttgtatctage atcttgc 1201tgggaaaatt grtattoc acscgttt cactgtcccs agagtcaca I -I-CI $eml. i mT-t stul toidyiluolput g. 0I,2 I.uni p. s uorlodklba thapi_ nmst iBk ca.dl.tCCC. -A . E.xbytd Kg. &PMp.ti .ATCT C>TCAT 0Pr ,.,k ,17 d eetcaf -23 positis 50cr4l.Q st Orsk l poi0m mth,,td lbas. eteto pim3 . th t turc'with C:. *TCTL[CXCCC]TCAT eA - Or at the so es ooro' Sten3 eS. d:*eg tldccc. .sze Ests10000 ei lo mtllr 1:. v-t' mol o too t mlt moo s01Ifo pSe Int ofTehol. Mascuet Massachusetts Institute of 'Technology u tsbolutl t Utt Uorto Ibhoti too slob todotoo coloaoutorttv -20 - 203- llo t .totYe Coto cerotl Bitcnlg V rcssEgneigCne Biotechnology Process Engineering Center - Try and keep the product melting temperature as high as possible, so that it is much greater than the melting temperature of the primer-dimers and genomic DNA products Keep the primer melting temperatures of the forward and reverse primers as close to > each other as possible > Software will give a list of candidate primers in ranked order as determined by their algorithm > Selectprimersfrom thegeneratedoptionsthat: > Avoid runs of 3 or more G/C at the 3' end > Avoid complementarities within the primer sequences and between the primer > Avoid complementarities and mismatches of 2 or more bases at the 3' ends of the primer and the target-template sequence > Avoid a 3' end T pair > Use primers that give a product with a high melting temperature > As far as possible, make sure to select a primer set that binds to a sequence on the gene that lies in the intron-exon boundary (this can be known by looking at where the primer binds to in the mRNA sequence and then comparing the sequence where the primer binds to with the complete cds sequence) Step 4: Verijy Targets of Ptimer Design > > Click on the 'short, Go to NCBI Blast (http://www.ncbi.nlm.nih.gov/BLAST/). nearly exact matches link' and input the determined primer sequence into the input box. Click on the BLAST tab and look up the set of genes that are listed each of which can be an RT-PCR product for chosen primer set. The primer sequence should yield the target gene with considerably greater significance than the next closest match for the same animal species (Rattusnorvegicus) Similarities between the gene specificity of different species is not important - 204 Massachusetts Institute of Technology MassachusettsInstitute of echology -204- Biotechnology Process Engineering Center~~~~~~~ Biotechnology Process Engineering Center Appendix 4 cDNA Preparationand Reverse TranscriptasePolymerase Chain Reaction (RT-PCR) protocols Material Needed: Deoxyribonuclease I (Invitrogen #18068-015), Omniscript Reverse Transcriptase Kit (Qiagen #205111), QuantiTect SYBR Green PCR Kit (Qiagen #204143), T7-Oligo(dT) 24 primer (Affymetrix #900375), RNase inhibitor (Ambion #2684) designed primers for genes of interest, RT-PCR grade water, DNA Engine Opticon, RNase-free tips, tubes, and 96-well plates, oligo-DTs and random hexamers (Qiagen, CA) Step l: Quantifiation of RNA concentration 1) Add 2 LLof RNA sample to 98 tL of DEPC-water (1:50 dilution) (Witrup UV requires 200 1 L of sample or 4 RNA:196 H 2 0O- use lower dilutions if there is too little RNA sample to give a good spec reading) 2) Blank UV Spec with DEPC water (or whatever the dilutant is) 3) Spec samples at 260 nm and 280 nm (NOTE - The UV spec in the Witrup Lab has much more precision and accuracy than Leona's) Determine quality (QRN,)and quantity (WRN)of sample: RNA WRNA= A260 QRNA , mLJ I.mpl ]ua,.or [pL] where aRNA =40 1.7 > QRNA> 2.1 A280 4) If the quality of the RNA sample falls outside of the defined range it should not be used. Step 2: DNase Treatmentof RNA 1) Add the following to an RNAse-free, 0.5 mL Eppendorf, on ice · 200 ng RNA sample * 1 iL 10X DNAse I reaction buffer Institute of Technology - 205 Massachusetts Magasachuseas Institute ofTechnology - - 205- Biotechnology Process Engineering Center~~~~~~~~~~~~~~~~~~~~ Biotechnology Process Engineering Center · 1 tzL DNAse · DEPC treated water to make upto 10 ~LL I, amplification grade, 1 (Note: reaction is optimized from 50ng-2[g of RNA. Regardless, of the initial amount of RNA be sure to perform the RT reaction at the same RNA concentration everytime to standardize experimental error) 2) Incubate tubes for 15 min at room temperature. 3) Inactivate the DNAse by adding 1 tL of 25 mM EDTA solution to the mixture. 4) Heat for 10 min. at 65"C. After that, keep the mixture on ice for use in the reverse transcription, or store at -80"C until its time for cDNA preparation. Step 3: ReverseTransciption:Total sample volume before the start of this step: 11 [zL 1) Make fresh dilution of T7 primers (or random hexamers) if 18s gene also needs to be transcribed to cDNA), and RNase inhibitor · RT buffer from 10x to x in RT-PCR H20 (1:10 dilution). · RNase Inhibitor (40 U/iL stock) to 10 U/xL in RT buffer (1:4 dilution) * T7 primer (50 FM stock) to 10 VtMin RT-PCR H2 0 (1:5 dilution) or random hexamers (dilute stock in RT-PCR water i.e. RNAse free water to 10 FM) 2) Prepare the master mix (everything in table below except the RNA template) on Ice (the values below are per 11 jiL solution of 200 ng of DNA'sed RNA). If random hexamers are being used, use same volume as oligo-DT in Master mix. CempN"a Find eo wi dk, Vdbmw/r.ckn Medw i I~ ki[fLerR1 pi 2 dT'F Mix rM Ix pi 0 eah dNTP CloIdT pirier 110 A' 2 0 pI ANaseInhiblr (IOur10 s/ p I O pi I Olriacrlpl I .0 pi d units ItpT20 pl r'ai a T ritxipwt ANR4o*ee vwa TenpIM I pM ulsper .n 2l re accr ciCl Va loble NA TemFkldeRNA cldd d d'p 5 Varible 1f_.dh,dl.. Technology Institute of Massachusetts Institute Massachusetts ofTechnology Up 2 pg r 20 p machord ,A iba.1 -- 11 - 206 206- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center 3) Add the appropriate amount of master mix - 9 .L to a 11 ZILsample of DNA'sed RNA i.e. 9p.L per 200 ng of starting RNA 4) Do make 2-3 samples of no-RT control using the RNA from some of the samples, ie. add 9 IlL of a special Master Mix solution that has all the components listed in the table above except the Omniscript Reverse Trasnscriptase. Instead of the 1 L Omniscript Reverse Transcriptase enzyme, use 1 L of DEPC/ RNAse free/ RT-PCR water in the special Master Mix.. The no-RT control can be used during the PCR step to see if all the genomic DNA has been digested by the DNAse step. Ideally we should see no amplification of the no-RT controls during PCR. 5) Centrifuge the samples with the master mix added at 2000 rpm, room temperature and then incubate @ 37"C for 60 min. After that time, store cDNA @-80"C (or -20°C) until PCR (NOTE: cDNA is much more stable than RNA, its often good to store cDNA than RNA for long time periods) Step 4: Quantification of cDNA concentration 1) Add 2 gIL of RNA sample to 98 pL of DEPC-water (1:50 dilution) (Witrup UV requires 200 jgLof sample or 4 RNA:196 H 2 0 - again use lower dilutions (more concentrated cDNA sample) if there is too little cDNA that a good spec reading is not possible. 2) Blank UV Spec with DEPC water (or whatever the dilutant is) 3) Spec samples at 260 nm and 280 nm (NOTE: The UV spec in the Witrup Lab has much more precision and accuracy than Leona's) 4) Determine quality (Qc,,,) = and quantity A L 1.8 > QNA ecDNA QDNA = A26 (WINA) facor 1000[I] > of sample: where soN A = 33 2.0 A 28 0 Technology Institute of Massachusetts Institute Massachusetts ofTechnology - 207 - - 207- Biotechnology Process Engineering Center Biotechnology Process Engineering Center 5) If the quality of the cDNA sample falls outside of the defined range it should not be used. Step 5: Real-Time PCR 1) Prepare master mix according to the following table (1.5 [.L of forward and reverse primers per 1 Figof cDNA, add that amount of RNAse free water to make the total reaction volume equal to 50 tL). The master mix is everything in the table below except the template cDNA. 2x WWTO V Gf..t.. 2S.. m MdW AMd' fimu d iner A Pltew Bm llpme ] Temd*ls DNAid ultlO IiL J.d odv 4J rl 0.3 m' d lO VM' 3 lid00 Totd*Am elon So 2) Aliquot master mix into white PCR tubes or plates (MJ Research, MA) 3) Add 1 Fdof the cDNA template to 49 Vl of the master mix. 4) Cover the plate/ tubes with clear caps (DON'T forget to do this!) 5) Program DNA Engine Opticon as follows. Line 3 should be changes depending on the annealing temperature of the designed primers. Do not run for more than 45 cycles. ' -- Prot toco . I P i: II I I iI _ 1. Incubate at 95.0 C for 00:15:00 2. Incubate at 94.0 C for 00:0015 3. Incubate at 57.0 C for 00:01:00 4. Incubate at 72.0 C for 00:00:30 5. Plate Read & Goto ine 2 for 45 moretimes 7. Incubate at 72.0 C for 00:02:00 8. MeltingCurvefrom 50.0 C to 98.0 C, read every 1.0 C, hold 00:00:01 END iI. f eholg nsiut Maschstt Massachusetts Institute of echnology -28 - 208- ioeh:lg Prcs nieigCne Biotechnology Process Engineering Center Appendix 5 Hepatocyte isolation and Spheroid Formation Hepatocytes were isolated from 150-230 g male Fischer rats with a modification of Seglen's two-step collagenase perfusion procedure [101] as described previously [47]. The resulting cell suspension was centrifuged three consecutive times at 50g (2 min each). Next, 21.6 ml of Percoll (Sigma-Aldrich, St. Louis, MO), was mixed with 2.4 ml of Hanks Balanced Salt Solution (HBSS, Sigma, MO), and added to 25 ml of the centrifuged cell suspension at a cell density of 5-7 million cells per milliliter of solution, as described in the literature [102]. The solution was further centrifuged at 50g for 10 minutes. Percoll isogradient centrifugation resulted in the separation of dead cells as well as a significant portion of the non-parenchymal cells in a floating top layer that was discarded. The sedimented hepatocytes were then resuspended in hepatocyte growth medium (HGM) [103], [47] but without hepatocyte growth factor (HGF). The final cell viability, as determined by trypan blue exclusion, was approximately 90-95%. Spheroidal cell aggregates were formed in suspension cultures similar to those described by Wu and co-workers [20] wherein one hundred milliliters of HGM was mixed with 30 million cells from the Percolled cell suspension solution in a 250-mL spinner flask (Bellco Glass, Vineland, NJ). The flasks were stirred at 85 rpm for up to 72 h. On the third day, spheroids of the desired size range (100 - 300 .un)[47]., were separated using appropriately sized filter meshes (Sefar America, Kansas City, MO) and re-suspended in 25 mL of rinse medium. The rinse solution comprised phenol red-free Dulbecco's modified Eagle's medium (DMEM) with sodium pyruvate (110 mg/mL) and glucose (1 g/L) (Life Technologies, Rockville, MD) supplemented with bovine serum albumin (2 g/L, Sigma, St. Louis, MO) and penicillin-streptomycin centrifuged at 4 0g (100 U/mL). The size-separated spheroids were for 3 min. The floating debris was then removed and the spheroid pellet was re-suspended in 30 mL of HGM. Massachusetts Institute Massachusetts Institute of of Technology Technology - 209 209- - Biotechnology Process Engineering Center Biotechnology Proess Engineering Center Appendix 6 Bioreactor Seeding and Maintenance of Culture imaging & spectroscopy vent filt The bioreactor system was primed with the rinse solution to passivate the bioreactor, connector and tubing surfaces, and to remove air bubbles from the flow paths. Prior to seeding, reservoir bottles were aspirated and refilled with 15 mL of HGM. For hydrodynamic seeding, a syringe filled with 1 ml cell-spheroid suspension was placed at the inlet of the upper chamber (port #1) and a second syringe containing -0.5 ml of PBS was placed at the bioreactor upper outlet (port #2). The bottom bioreactor outlet (port #3) was unclamped by removing the tubing from the peristaltic pump rotor and the bioreactor was tilted approximately 450 with the bioreactor outlet (port #2) higher than the inlet (port #1). The cell-spheroid suspension was manually injected into the upper chamber at the flow rate of -0.5 ml/min. The spheroids entered into the scaffold channels through a combination of settling and flow of cell culture medium from the upper chamber into the lower chamber aided by the resistance of the syringe on the bioreactor upper outlet (port #2). -210Instimte of Technology Massachusetts Massachusetts Institute ofTechnology -210- BiotechnologyProcess EngineeringCenter~~~~~~~ Biotechnology Process Engineering Center Following seeding, cell culture medium was pumped through the upper bioreactor chamber at 0.5 ml/min (with pump A) and collected in a waste container for one minute to clear cells from the top surface of the scaffold. The upper recycle tubing was then reconnected to the bioreactor (port #2). At this time, the bottom tubing was inserted into the peristaltic pump B and the perfusion flow rate (cross-flow) down through the seeded channels was set to a desired flow rate. The selection of flow rates was based mainly on physiological shear stress conditions and mass transfer considerations (Chapter 3). After one hour, cell culture medium in the reservoir was replaced with 15 ml of fresh medium to reduce residual cells/debris trapped in the circulation loop. Spheroids seeded into the channels are initially held in place by the membrane or filter, and after initial, attachment and reorganization (-1 day), by adhesion to the collagen-coated channel walls. During the "top down" perfusion, the residual cell debris is likely to gradually dog the micro porous filter. To maintain a constant perfusion rate and keep the filter in the bioreactor free of debris, 24 hours after the seeding we placed an inline filter between pump B and the reservoir, and reversed the direction of the cross-flow while maintaining the same value of the perfusion or crossflow rate. We kept this direction of the cross-flow for the duration of the experiment. For five-micron pore-size DURAPORE filter (Millipore Corp., Bedford, MA) in the bioreactor, we used a 0.8/0.2-pm pore-size double layer inline syringe filter (Pall Corporation, Ann Arbor, MI). In this way, cell debris capable of clogging the filter in the reactor was captured in the inline filter. By replacing the inline filter every 72 hours, a constant "bottom up" cross-flow was maintained through the channels with cells. In case of the control "zero-cross-flow" bioreactors, 24 hours after seeding the bottom support silicon scaffold with channels was removed and replaced by a solid, 230 Vtm thick polycarbonate piece. The reason for using the solid polycarbonate piece was to drastically reduce gas exchange between cell culture medium in the bottom reactor chamber and the cells in the upper scaffold. Therefore, the cross-flow pump B was used only in the bottom-down flow direction immediately following the seeding and was disconnected when the blocking lower scaffold was installed. Cell medium was changed every 3 days by replacing the reservoir with a new one containing 15 mL of fresh HGM. nttt ofTcnlg : Mascuet Massachusetts Institut~e of Technology 21 -211 - itcnloyPoesEgneigCne Biotechnology Process Engineering Center Appendix 7 Measurement of the total number of viable cells in cultures using the measured amount of total RNA and RT-PCR against the 18s gene Total RNA isolated from the cells in the 2D and 3D cultures was used to quantitate the total number of viable cells in the culture and the average viable cell number per channel in the scaffold (in case of the 3D bioreactor culture). Total RNA was isolated and purified from the cultures as described in Appendix 2. cDNA was prepared from the total RNA as described in Appendix 4 and RT-PCR against the 18s gene (designed as described in Appendix 3) was performed as described in Appendix 4. In order to determine the unknown viable cell number in the scaffolds, the total RNA from a number of samples of Percoll-separated hepatocytes of known cell densities were isolated. Total number of viable cells in a bioreactor was determined from the total RNA versus number of isolated hepatocytes standard curve (similar to Figure 67 in Chapter 6). The number so determined was validated using the 18s standard curve plotted using C,r values generated from RT-PCR reactions against the 18s gene done on all the cell standard (similar to Figure 6-6, in Chapter 6). Technology Institute of Massachusetts Institute Massachusetts ofTechnology -212- -212 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center - Appendix 8 Preparationof 2D collagen gel sandwich cultures Two dimensional collagen sandwich cultures were prepared similar to previous methods suggested by Dunn and co-workers and later reviewed extensively in literature [18, 44, 122, 194]. 35 mm tissue culture treated six-well plates (Falcon, USA), were coated with 600 ul of a collagen mixture (4.7 ml sterile water, 1.3 ml of modified PBS solution [g glucose, 1.85g sodium bicarbonate, in 50 mL PBS solution], 6.9 ml of a 3mg/ml collagen (type I) solution (Vitrogen, USA)), and with pH of solution adjusted to - 7.4. Plates were coated overnight to allow the collagen to gel, in an 8.5% C02, 37°C environment. Percolled primary rat hepatocytes were seeded at a density of 50,000 cells/! cm2, and evenly distributed on the plate, in lmL of Hepatocyte Growth Medium (HGM). Following an overnight incubation at 37'C and 8.5%C02, that allowed the cells to attach to the substratum, the media was aspirated from the plate and 300 ul of the collagen mixture was added to form the second layer of the sandwich. The collagen was allowed to gel over a 45 -60 min period, following which 1 mL of HGM was added to the well. The cultures were incubated in a controlled 37°C, 8.5%C02 environment. Media was changed everyday. nttt fTcnlg Mascuet Massachustts Institute ofTechnology 23-BoehooyPoesEgneigCne -213- Biotechnology: Process Engineering Center Appendix 9 Preparationof 3D spheroid and 3D Matrigel spheroid cultures 30 million percolled isolated primary rat hepatocytes were added to 100 mL of HGM in a 500 mL spinner flask (BellCo, NJ, USA) on a spinner table set at 85 rpm [20]. The cells aggregated to form spheroids. Flasks were taken down on Day 2 and Day 3 after cell seeding, for RNA extraction. Day 3 or Day 4 spheroids were used to seed the bioreactors. BD Biocoat'M Matrigel Matrix 6-well Multiwell plates were used. The Matrigel plates were kept frozen at -20° until the day of use. On the day when cells are to be seeded on the plates, the multiwell plate is brought to a temperature of 4°C over a twio hour incubation period. The plates are then placed in a tissue culture laminar hood for 30 min. Next the plates are transferred to an incubator at 37°C and 8.5%CO2 for a period of one-hour. Percolled hepatocytes are added to the plates at a density of 80,000 cells/cm2 (As suggested in supplier's optimized protocol). 2mL of HGM media are added to each well after the addition of the hepatocytes. Medium was changed every other day, as suggested by the supplier. Within three days the cells forms into spheroids, and the cultures are taken down after 7, 13 and 20 days after cell seeding. =nttt Massachusetts~ ofTehooy-1-BoehooyPoesEgneigCne - 214- Massachusetts Institute of Technology Biotechnology Process Engineering Center Appendix 10 Induction experiments on 2D collagen sandwich and 3D microreactor cultures Media was changed in both the 2D and 3D cultures everyday, and prototypical inducers were added on Day 4 after cell seeding. Control cultures that did not receive the inducers, were treated with an equal volume of the solvent in which the inducers were formulated (e.g. DMSO for 3MC, HGM for CLO and DMSO for PCN). Cultures were taken down on Day 7 for RNA extraction. Inducer concentrations that produced maximal induction in 2DCSW cultures relative to basal were used - 10 FiMPCN, 5 FiM3MC, 100 1tMCLO. Technology ofTechnology Institule of Massachusetts Instituie Massachusetts -215- -215 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendix 11 Accession numbers for mRNA and complete cds sequences used The specific mRNA or cDNA sequences obtained from the NCBI database accessible at http://www.ncbinlm.nih.gov/entrez/query. fcgi?db=Nucleotide was used to design primers using the primer3 software at the Whitehead bin/primer3/primer3_www.cgi). Institute (http://frodo.wi.mitedu/cgi- Specific accession numbers used include X63410 - Hydroxysteroid Sulfotransferase, IK01931- GST-Ya, M13506 - UDPGT, NM_022180 - HNF4a, NM_012742 - HNF3a, XM_343991 - HNF1P, NM_012669 - HNFl, NM_012524 - CEBPI. NM_024125 - CEBPa, AF082126 - AHR, AB105071 - CAR, AF151377 - PXR, NM_013196 - PPARlX, U18374 - FXR, NM_012805 - RXRoc L00319 CYP2B1, XM_2152557 - CYP2C6, U33173 - CYP2C11, X64401 - CYP3A1, NM_153312 CYP3A2, NM_031543 - CYP2E1, NM_012541 - CYP1A2, NM_175837 - CYP4A1, NM_012942 - CYP7A1, NM_131906 - OATP2 (or Slc21a5), AJ277827 - Hif3a, CYP7A1 J02722 - Heme Oxygenase-1, X01117 - 18s housekeeping gene. Technology Massachusetts Massachusetts Institute Institute of ofTechnology -216- -216- Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendix 12 Primer sequences used in RT-PCR studies Oligonucleotide primers for CTTCAGTTCCAAGGCCAAG-3 Hydroxysteroid forward and reverse), UDPGT CATCA-3 forward and reverse), HNF1P forward and 5- reverse), HNF-4a (5-CTGAGACTCCACAGC 5-CTAGATGGCTTCCTGCTTGG-3 TCAACGCTTGT'rCGTCAAG-3 forward and reverse), forward and 5-CTL'CAAC GCGTCTGTACCA-3 reverse), HNFlat 5-GCCATCTGGGTGGAGATAAA-3 reverse), reverse), CEBPO (5-ACTTCA GCCCCTACCTGGAG-3 forward and 5-GGCTCACGTAACCGTAGTCG-3 forward and reverse), CAR (5-GGAGGACCAGATCTCCCTTC-3 ATCITGT-3 GTTTCATGGCCCTTCTGAAA-3 forward and forward and 5-GACCGCATCTITCC reverse), FXR forward and 5-GCCTCTGCTCGATGTCCTAC-3 GTCAAGCAGCAGACAAGCAG-3 and ATGGACT-3 reverse), AG-3 forward and 5-GGAAGTGTTC CYP2C11 forward (5-CAAGTACCAC forward and 5-GTGGGGACAAGGTCAATATATCTC-3 reverse), CYP3A1 (5-CATCAGAGGCCCAGCTAGAG-3 forward and 5-AGGACCCAGGT TITCCAGTGT-3 reverse), CYP3A2 (5-GTAGTACTCTTTCCATTCCTCACCC-3 Technology of Technology Institute of Massachusetts Institute Massachusetts 5- reverse), RXRa (5- reverse), CYP2C6 (5-TGGGGAAAAGGAACATGAG-3 5-GGATAAACGTGGGGTCACAG-3 TCTGAGGT and (5-ATGTCCGGA forward and 5-GAGAAGGAGGCAATCAGC reverse), CYP2B1 (5-CAACCCTTGATGACCGCAGT-3 AGGATrGGAAGC-3 forward reverse), PPARa (5-TCACACAATGCA ATCCGTfT-3 5-GCCAGAGATTTGAGGTCTGC-3 GTTCTGTCAGG-3 reverse), AHR 5-AITCATrGCCAGGAAACCAG-3 (5-GGTCTTCAAATCTGCCGTGT-3 reverse), PXR forward and CEBPa (5-GCCAAGAAGTCGGTGGA TAA-3 forward and 5-CGGTCATTGTCACTGGTCAA-3 (5-ACTACACGCCAGACCAGCTT HNF3a (5- 5-TCACACTTGAAGCGCT1lTG-3 (5- CGTAGCAAGGAACTCCCAAG-3 5-ACCAGTCCCACAGTGTCCTC-3 (5- forward and 5-GTITTGCATCCAT (5-GCCTTCTCACTGCCTIGAAG-3 GTCTTACGGCAACAAAAGAGGCAG-3 (HSST) 5-GTGAAGTGAITCTTCCAGTC-3 reverse), GST-Ya (5-CTGGCAAAAGACAGGACC-3 GGGAAGC-3 Sulfotransferase 217 -217- forward Biotechnology Process Engineering Center Biotechnology Process Engineering Center and CYP1A2 (5-TATCTACG forward and 5-CTCTATGAGGAGACAGTCAGTCACA-3 ATACCTACCTGGAAGCC-3 reverse), CYP2E1 reverse), 5-GGTGCTTATGCTTAGAATCCAGAC-3 5- and forward (5-GGGGAAGAACCCACACCTAT-3 GTCATCCCCCTGCTTCACTA-3 reverse), CYP4A1 (5-TCATGAAGTGTGCCTTCAGC-3 forward and 5-GATGTTCCTCACACGG AAGAG GCTAAGACCGCCTTC-3 forward and 5-CCTCTGGCGAAGAAACTCTG-3 reverse, Hypoxia Inducible Factor GAGAGG-3 forward and AGAACGGGTTGATITC-3 OATP2 3-alpha reverse), (HiF-3x) - (5-TCACATACTGCGAC reverse, 5-TGCTGACTGCATCAGAGTCC-3 forward and 5-AAAAC (5-CAATTCGGTATCCCCACATC-3 ACCAATC-3 Heme Oxygenase I (5- GAGT-3 reverse), 18s (5-TTTGG GTGACCATGCTTCC-3 forward and reverse), 5-CTGCACATCCAAC (5-GCAATTATTCCCCATGAACG-3 forward and 5- GGCCTCACTAAACCATCCAA-3 reverse) were designed using the Primer3 open source bin/primer3/primer3_www.cgi) software (website link: http://frodo.wi.mit.edu/cgi and synthesized by Qiagen, CA. Massachusetts~~ Massachusetts Institute ofTechnology ~~~ Inttt -218 of Tehooy-1-BoehooyPoesEgneigCne Biotechnology Process Engineering Center Appendix 13 Melting and annealing temperatures of designed primers Gene OptnimumProduct Melting Optimum Annealing Temperature (C) Temperature (C) P450's - Phase I Metabolism Genes CYP 1A2 51 83 Not optimized Not optimized 55 81 51 79 55 81 CYP 2E1 55 78 CYP 3A1 CYP 3A2 59 80 55 75 CYP 4A1 55 79 CYP 4A3 55 79 CYP 7A1 51 '77 CYP CYP CYP CYP 2A1 2B1 2C6 2C11 Phase II Metabolism Genes GST-Ya UGT ST-1A1 -~~~~II.i.54 82 58 83 59 79 IE I"--I"II~I-II--~~"~YIWIIU·U·C.·-- ii 11r~lrIIIIIIE~lI I i 111111 iII IlI IlElIIIIlIEl - - I --- i.E IE- II- --- -l - - · IIIYIII_·1L -l -- ~~~~~~~"p""e"""""C"II-~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~ 3*JIN ~C -- Es'-'.0h.atti^.c.1. ------- ----- ------ 1~9*r3 W~C~')Z:~ IrF~~~~Yw vya~~~~~~~~~t ' -r - -, - --- r--. Stress/ proliferation markers p 53 Chkl Prion institute .fof Technology Niassachuserts Manssachusetts Intitite echnoloyn 5') 82 59 82 Not optinmized --219219- Not optimized Biotechnology Process Engineering Center Biotechnologyy Process nginering Center I I ... MM" ~ vfi-4-. v.Xk'.: - ~ mm'salffilm 1= MINAM 111.l~b~ aU - r·· .·- - -- I.,...."··~-. .. . f K ~' MONTE. .14'20 Oll'' : I''I AI ,I' , I~ ;~ti - ·_1. .i i.' .. f:f: . . . f-. .... .-.. . .- ...--. :. ~@tMt-, V, wrig ~sli I I t. ,., 04.- ~~f~i~~t~s~~e~ ~~~~~~~ii~~~~~~~~~~~~~~~~~~~t~~~~~~~~~~~ . IFE a Ii M1Uit rw! A111 t I I _I BSt# 50-63 MDR-2 OATP-2 . · . 53 m~1 53 79 _ 76 76 Intuco ehooy-2-BochoovPoesEgneigCne Massachusetts~A ,asschusetts Instimiteof echnolog -220- Biotcchnolop, Process E ineer4g Center Appendix 14 MicroarrayProcessing The Affymetrix small sample labeling protocol (www.affymetrix.com) was used for all total RNA samples to generate labeled cRNA. Labeled samples were hybridized in triplicate to the Rat Genome Array (U34A) Gene Chips. Data from arrays were processed using nonlinear normalization approach through DNA-Chip Analyzer version 1.2. The average and the standard deviation of replicates were calculated and probes that were "Absent" throughout the data set were eliminated from further analysis. Fold changes were calculated by dividing the mean of replicates of the culture condition by that of intact liver tissue for each gene. Quality filtering using coefficient of variation (CV) was performed to data with high variability. Using S-Plus software (Mathsoft, Cambridge, MA), a 95% CV quantile was calculated for each sample. Significance was assigned for samples within 95% CV threshold and samples with high variability eliminated. The number of probe sets remaining after filtering was 2098 probes (23.84%). More detailed microarray protocols are available elsewhere [130]. Technology Massachusetts Institute ,%Wsachusetts Institurte of ofTechnology -221- -221 - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendix 15 Testosterone Metabolism studies Fresh albumin-free Hepatocyte Growth Media (HGM) was added to both the 2D Collagen sandwich (2DCSW) as well as the 3DB (3D Bioreactor) cultures. The 2DCSW cultures were shaken gently on a shaker kept inside an incubator that provided a controlled 37°C, 8.5%C02 environment. Media was changed twice in the 2DCSW at 30 minute intervals to help wash out the albumin present in the collagen. Media was changed twice, again at 30 min intervals, in the 3DB to clean out the rat albumin present in the system. 250 jlM concentration of testosterone was made up in fresh albumin-free media solution, and was added to the 2DCSW cultures as well as the 3DB. Products of metabolism of testosterone were collected 60 min. after the addition of the substrate to both the 3DB and the 2DCSW cultures. The use of the shaker with the 2DCSW helped eliminate concentration gradients in the media. Linearity of the Michaelis-Menten kinetics was verified over this time-span (data not shown). To the samples from the 2DCSW and 3DB cultures, a known concentration of an internal standard (Methyl Testosterone) was added. The samples were them vacuum dried (SpeedVac, Thermo Electron Corporation, MA) and testosterone and the hydroxy-products of testosterone were extracted from the dry powder using methanol as solvent. Quantification of the concentrations of testosterone and the products of metabolism of testosterone was done by HPLC analysis on an Agilent 1100 series (Agilent Technologies, Waldbronn, Germany) which consisted of a 9725 Rheodyne injector (Rheodyne, Rohnert Park, CA) and a G1314A variable wavelength UV detector at 240 nm (Agilent Technolgies). An aliquiot of the extracted sample (5uL) from each incubation was injected on a Capcell Pak C18 column type UG120 size 2.0mmID X 150mm, 5um particle size; Shiseido Fine Chemicals, Tokyo, Japan and eluted at a flow rate of 200ul/minute by the gradient with the mobile phase, which consisted of solvent A (20 mM ammonium acetate in 10% methanol) and solvent B (90% methanol). The solvent gradient (solvent B) used for eluting testosterone and its metabolites was as follows: 0 min, 10%; 0-10min, 10%10-20min, 30%; 20-30min, 55%; 30-38 min, 55%; 38-45 min, 100%; 45-50 100%. The gradient was then returned to the initial condition (10% B) and held for 8 Technology ofTechnology Institute of Massachusetts Institute - 222 222- - Biotechnology Process Engineering Center Biotechnology Process Enginieering Center minutes before analysis of the next sample. A standard curve of the ratio of AUC units of testosterone (or products of its metabolism) to that of the internal standard, was plotted against known concentrations of the standards. Standards were all purchased from Steraloids, RI. Techno1o' Massachusetts Mlassachusetts Insfitute Institute of of Technology - 223 223- - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Appendix 16 The Drug Development Process - a detailed overview Synthesis / The following sequence of research activities begins the process that results in development of new drugs: 1) Target Identification: Drugs usually act on either cellular or genetic chemicals in the body -- known as targets -- which are believed to be associated with the disease. Scientists use a variety of techniques to identify and isolate a target and learn more about its functions and how these influence disease. Compounds are then identified, which have various interactions with drug targets that are helpful in treatment of a specific disease Massachusetts~~ Massachusetts Institute of echnology 2 ~~~ Inttt - 224- ofTcnlg itcnloyPoesEgneigCne Biotechnology Process Engineering Center 2) Target prioritization/validation: Each drug target is compared to others based on their association with a specific disease and their ability to regulate biological and chemical compounds in the body -- in order to select targets most likely to be useful in the development of new treatments for disease. Tests are conducted to confirm that interactions with the drug target are associated with a desired change in the behavior of diseased cells. This helps identify compounds that have an effect on the target selected. 3) Lead identification: A lead compound or substance is one that is believed to have potential to treat disease. Laboratory scientists can compare known substances with new compounds to determine their likelihood of success. Leads are sometimes developed as collections -- known as libraries -- of individual molecules that possess properties needed in a new drug. Testing is then done on each of these molecules to confirm its effect on the drug target. 4) Lead optimization: Lead optimization compares the properties of various lead compounds and provides information to help select the compound or compounds with the greatest potential to be developed into safe and effective medicines. Often during this same stage of development lead prioritization studies are conducted in living organisms (in tvio) and in cells outside the organism (in vitro)to compare various lead compounds and understand how they are metabolized and affect the body. What is required before an investigational drug can be tested in human volunteers? 5) In the preclinical stage of drug development an investigational drug must be tested extensively in the laboratory to ensure that it will be safe to administer to humans. Testing at this stage can take from 1 to 5 years and must provide information about the pharmaceutical composition of the drug, its safety, how the drug will be formulated and manufactured, and how it will be administered to the first human subjects. Results of all testing must be provided -- to the Food and Drug Administration (FDA) in the U.S. and/or other appropriate MascuetIn-ueo ehooy-25-BoehooyPoesEgneigCne -225- Miassachusets Institute ofTechnology Biotechnology- Process Engineering Center regulatory agencies -- in order to obtain permission to begin clinical testing in humans. Regulatory agencies review the specific tests and documentation that are required to proceed to the next stage of development. 6) Setting up Chemical Manufacturing Controls (CMC): The results of preclinical testing are used to determine methods to formulate the drug for its intended clinical use. For example, a drug that is intended to act on the sinuses may be formulated as a time-release capsule or as a nasal spray. Regulatory agencies require testing that documents the characteristics -- chemical composition, purity, quality and potency -- of the drug's active ingredient and of the formulated drug. 7) Pharmacology/Toxicology studies: Pharmacological testing determines effects of the candidate drug on the body using a series of screens that determine the effects of the new chemical entity on the p450's. Toxicology studies are also conducted to ensure that any risks to humans are identified. How are investigational drugs tested in humans? 8) Testing of an investigational new drug begins with submission of information about the drug and application for permission to begin administration to healthy volunteers or patients. Investigational New Drug (IND)/Clinical Trial Exception (CTX)/Clinical Trial Authorization (CTA) applications. INDs (in the U.S.), CTXs (in the U.K.) and CTAs (in Australia) are examples of requests submitted to appropriate regulatory authorities for permission to conduct investigational research. This research can include testing of a new dosage form or new use of a drug already approved to be marketed. In addition to obtaining permission from appropriate regulatory authorities, an institutional or independent review board (IRB) or ethical advisory board must approve the protocol for testing as well as the informed consent documents that volunteers sign prior to participating in a clinical study. An IRB is an independent committee of physicians, community advocates and others that ensures that a clinical trial is ethical and that the rights of study participants are protected. Clinical testing is usually described as Technology ofTechnology Massachusetts Massachusetts Institute Institute of 226 -226 - - Biotechnology Process Engineering Center Biotechnoloy Process Eineering Center consisting of Phase I, Phase II and Phase III clinical studies. In each successive phase increasing numbers of patients are tested. Phase I Clinical Studies: Phase I studies are designed to verify safety and tolerability of the candidate drug in humans and typically take 6 to 9 months. These are the first studies conducted in humans. A small number of subjects, usually from 20 to 100 healthy volunteers, take the investigational drug for short periods of time. Testing includes observation and careful documentation of how the drug acts in the body -- how it is absorbed, distributed, metabolized and excreted. Phase II Clinical Studies: .Phase II studies are designed to determine effectiveness and further study the safety of the candidate drug in humans. Depending upon the type of investigational drug and the condition it treats this phase of development generally takes from 6 months up to 3 years. Testing is conducted with up to several hundred patients suffering from the condition the investigational drug is designed to treat. This testing determines safety and effectiveness of the drug in treating the condition and establishes the minimum and maximum effective dose. Most Phase II clinical trials are randomized -- randomly divided into groups, one of which receives the investigational drug, one of which gets a placebo containing no medication, and sometimes a third that receives a current standard treatment to which the new investigational drug will be compared. In addition, most Phase II studies are doubleblinded -- meaning that neither patients nor researchers evaluating the compound know who is receiving the investigational drug or placebo. Phase III Clinical Studies: Phase III studies provide expanded testing of effectiveness and safety of an investigational drug, usually in randomized and blinded clinical trials. Depending upon the type of drug candidate and the condition it treats this phase of development usually requires one to four years of testing. In Phase III, safety and efficacy testing is conducted with several hundred to thousands of volunteer patients suffering from the condition the investigational drug treats. Technology Massachusetts MassachusettsInstitute Institute of ofTechnology 227 -227 - - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center Authorization Application (MAA) NDAs 9) New Drug Application (NDA)/Marketing (in the U.S.) and MAAs (in the U.K.) are examples of applications to market a new drug. Such applications document safety and efficacy of the investigational drug and contain all the information collected during the drug development process. At the conclusion of successful preclinical and clinical testing, this series of documents is submitted to the Food and Drug Administration (FDA) in the U.S. or to the applicable regulatory authorities in other countries. The application must present substantial evidence that the drug will have the effect it is represented to have when people use it or under the conditions for which it is prescribed, recommended or suggested in the labeling. Obtaining approval to market a new drug frequently takes between 6 months and 2 years. Does testing continue after a new drug is approved? After the FDA (or other regulatory agency for drugs marketed outside the U.S.) approves a new drug, pharmaceutical companies may conduct additional studies, including Phase IIIb and Phase IV studies. Late-stage drug development studies of approved, marketed drugs may continue from several months to several years. Phase IIIb/IV Studies: Phase IIIb trials, which often begin before approval, may supplement or complete earlier trials by providing additional safety data or they may test the approved drug for additional conditions for which it may prove useful. Phase IV studies expand testing of a proven drug to broader patient populations and compare the long-term effectiveness and/or cost of the drug to other marketed drugs available to treat the same condition. Post-Marketing Studies: Post-marketing studies test a marketed drug in new age groups or patient types. Some studies focus on previously unknown side effects or related risk factors. As with all stages of drug development testing, the purpose is to ensure the safety and effectiveness of marketed drugs. Technology ofTechnology Massachusetts Institute Institute of 228 -228 - - Biotechnology Process Engineering Center Biotechnology Process Engineering Center