CHEMICAL AND ELECTRICAL STIMULATION IN ENHANCING HUMAN NEURAL STEM CELL MIGRATION A Project Presented to the faculty of the Department of Biological Sciences California State University, Sacramento Submitted in partial satisfaction of the requirements of the degree of MASTER OF ARTS in Biological Sciences (Stem Cell) by Michelle So SPRING 2012 ii © 2012 Michelle So ALL RIGHTS RESERVED ii CHEMICAL AND ELECTRICAL STIMULATION IN ENHANCING HUMAN NEURAL STEM CELL MIGRATION A Project By Michelle So Approved by: _____________________________________, Committee Chair Thomas Landerholm, Ph.D. _____________________________________, Second Reader Jan Nolta, Ph.D. _____________________________________, Third Reader Christine Kirvan, Ph.D. _____________________________________ Date iii Student: Michelle So I certify that this student has met the requirements for format contained in the University format manual, and that this project is suitable for shelving in the Library and credit is to be awarded for the project. _______________________________, Graduate Coordinator Ronald M. Coleman, Ph.D. Department of Biological Sciences iv ___________________ Date Abstract of CHEMICAL AND ELECTRICAL STIMULATION IN ENHANCING HUMAN NEURAL STEM CELL MIGRATION By Michelle So Studies of multipotent neural stem cell (NSC) transplantations demonstrate their potential as a regenerative therapy to improve functional recovery following neurotrauma. However a major obstacle hindering greater tissue regeneration is the inefficient rate of engraftment and migration of transplant cells to the site of injury. Towards this challenge of improving NSC migration, other studies have discovered chemotactic or motogenic properties of certain trophic cytokines that are important for maintaining NSC multipotency. However fewer studies have investigated the use of physical migration cues, specifically direct current electric fields (EF) and even fewer look at the migratory responses of a combined chemical-electrical migration stimulation condition. In this study we compared the migration responses of human embryonic stem cell (ESC) line, H9, derived NSCs to chemical and electrical migration stimulation combinations in vitro. Our results suggests that trophic cytokines such as basic fibroblast growth factor (bFGF), epidermal growth factor (EGF) and adenosine triphosphosphate (ATP) traditionally known to induce a chemotactic response, did not have a positive chemokinetic effect on our H9-NSC migration. Conversely, exposure to direct current electric field stimulation enhances migration by increasing motility, migration directionality and distance travelled in a linear, less tortuous path. We also observed preliminary data that suggests contrary to intuitive expectations, v electrical and EGF combined stimulation did not additively increase cell motility significantly or for any extended period of time. Interestingly, the co-stimulation appears to increase directedness of electrotactic migration. This suggests that other motogenic cytokines and electrical co-stimulation may positively enhance hNSC motility and directionality in vitro. It also suggests the combination could have similar effect on NSC migration if applied in vivo and warrants further study towards optimizing stimulation conditions. However because EF stimulation is the major contributing factor behind the increase in motility and linearity of migration path, the addition of a chemical component to optimize electrotaxis must be carefully considered due to complicated regulation behind receptor activation and downstream intracellular cascade signaling. ____________________________, Committee Chair Thomas Landerholm, Ph.D. ____________________________ Date vi ACKNOWLEDGEMENTS There are few privileged scholars who have the luxury of opportunity, mentorship and security. I am honored to have been given the unique opportunity by Bridges to Stem Cells Program, the UC Davis Institute for Regenerative Cures, CSU Sacramento Department of Biological Sciences, and California Institute of Regenerative Medicine to study stem cell biology. It is because of these institutions that I can participate alongside a community of biological researchers and doctors striving to expand our understanding and capacity to promote the foundations and practice of human health and well-being. For their profoundly encouraging support and mentorship, I am immensely grateful to Drs. Min Zhao, Jan Nolta, Gerhard Bauer, Jing Liu, and members of the Zhao lab, notably Drs. Junfeng Feng, Yao Hui Sun, and Lei Zhang. From the CSUS family, I am indebted to Drs. Thomas Peavy, Thomas Landerholm, Christine Kirvan, and Susan Lindgren for their guidance. And though hardly ever given the full credit they deserve, I would like to express my deep gratitude to my family and friends who provide me the security and peace of mind in knowing that I am loved and in their thoughts. To my mother, father and sister, you’ve been right all along. And for all others who were involved in bringing this project into fruition, thank you, especially Brian Fury and Gregorio Gutierrez, who I expect to address as Drs in the near future. This work bears my name but it carries the work of all these giants. vii TABLE OF CONTENTS Page Acknowledgements ...................................................................................................................... vii List of Tables ..................................................................................................................................x List of Figures ............................................................................................................................... xi INTRODUCTION ..........................................................................................................................1 MATERIALS & METHODS .......................................................................................................13 Culturing H9-ESC Derived NSC for Migration Analysis.........................................................13 Chemical stimulation of NSC migration and motility ..............................................................14 Electric field (EF) stimulation of NSC migration and motility.................................................15 a) Chamber Preparation ............................................................................................................15 b) Application of Direct current EF Stimulation to hNSCs ......................................................17 Time lapse Recording of NSC Migration Behavior..................................................................19 Quantification of NSC Migration Parameters with Stimulation Modalities .............................19 Statistical Analysis of Migration Parameters ............................................................................19 RESULTS .....................................................................................................................................20 H9 hESC derived NSCs display characteristic designation and markers of NSCs...................20 hNSCs display motile but non-linear, highly tortuous random migration behavior ................21 Chemical stimulation alone shows no significant change in cell motility ................................23 Electrical stimulation significantly increases hNSC motility and directionality of migration..29 Chemical and electrical co-stimulation increases hNSC directedness but not motility consistently ..............................................................................................................................34 viii DISCUSSION ...............................................................................................................................39 Literature Cited .............................................................................................................................47 ix LIST OF TABLES Tables Page Table 1 Values of significance between NSC track speeds with chemical stimulation ................ 28 Table 2 Values of significance between NSC speeds and directedness with electrical stimulation. ......................................................................................................................................... 33 Table 3 Values of significance between NSC speed, directedness and distance when comparing electrical stimulation with chemical-electrical co-stimulation. ....................................... 37 Table 4 Values of significance comparing net displacement of EF only to chemical-electrical costimulation.. ..................................................................................................................... 38 x LIST OF FIGURES Figures Page Figure 1 Mechanism generating electric field at wound site. ......................................................... 9 Figure 2 Construction of EF Chamber.. ........................................................................................ 16 Figure 3 Electrotaxis Experiment Set Up.. ................................................................................... 18 Figure 4 H9 NSC exhibit motile phenotype in absence of stimulation......................................... 21 Figure 5 H9 NSCs display an actively motile but non-linear random migration in a heterogeneous population.. ............................................................................................. 23 Figure 6 hNSCs speed stable for several hours but inefficient due to tortuous random behavior 24 Figure 7 Chemical stimulation of growth factor-starved hNSCs with bFGF and extracellular ATP shows no increase in chemokinetic migration.. ..................................................... 26 Figure 8 Chemical stimulation of growth factor-starved hNSCs shows no increase in chemokinetic migration in EGF stimulation. ................................................................. 27 Figure 9 NSC motility is enhanced when exposed to electrical field. .......................................... 30 Figure 10 hNSC migration is less tortuous and more directed towards cathode when exposed to electrical field. ................................................................................................................ 31 Figure 11 hNSC migration directedness is quantified showing greater directional migration towards cathode when exposed to electrical field. ......................................................... 32 Figure 12 EGF combined with EF stimulation increases migration directionality but does not consistently increase motility. ........................................................................................ 35 Figure 13 Trends shows greater net horizontal displacement in EGF combined with EF stimulation. Greater cathodal migration compared to just EF alone. ............................. 36 xi 1 INTRODUCTION Acute traumatic brain injury (TBI) is the leading cause of long term disability in adults under 40, affecting 150 to 200 per million people globally (Fleminger and Ponsford, 2005). In the US alone, an estimated 1.7 million medical cases were reported annually from 2002 to 2006, resulting in 52,000 deaths (Faul, 2010). Cases vary widely and depending on the location and extent of injury to the brain, any number of physiological functions, bodily controls or cognitive functions could be permanently altered or lost. In many ways, TBI is an extremely expensive condition. Aside from the cost of years of physical therapy or in-home care, is the cost of permanent loss of full physical capacity and personal autonomy. But perhaps more devastating is the loss of cognitive functions such as critical thinking, reasoning, memory, language, sensory perception, and emotional intelligence, many functions we as a society consider to be foundations of being uniquely human. As a corollary, many patients must endure the psychological tolls of coping with long term disability and the changes to how they and society now perceives them. Additionally, as a nation, the direct and indirect fiscal cost of TBI injuries amounts to approximately 60 billion dollars in hospitalization, emergency treatments, post injury care, therapy and loss of occupational productivity (Finkelstein, 2006). For patients with the means to access resources, a multi-facetted treatment plan can be crafted involving a combination of surgical intervention, routine physical/occupational therapy and psychological counseling. Though a variety of these post-injury interventions are available, the recovery plan largely remains one of a rehabilitative strategy to assist endogenous recovery and retraining rather than a strategy of regeneration. Therefore, the long term outcome of such injuries to the central nervous system depends in large part on the age, general health and the capacity for 2 neurogenesis and regeneration in the individual (Faul, 2010). Coincidentally, the highest rate of TBI-related fatalities falls primarily in the elderly population (Faul, 2010). In children and young adults, we see predominantly long-term or permanent physical and cognitive handicaps (Faul 2010). Therefore there is a pressing need to improve our current therapies to produce greater functional outcomes and actual regenerative tissue repair following TBI. In traumatic brain injury, immediately after trauma, an acute inflammatory response recruits innate immune system components and astroglial cells to the lesion site. They phagocytose cellular debris, migrate towards damaged tissue and quarantine it by creating a barrier of glial cells (Richardson, 2010, Namas, 2009, Lo, 2010). The proliferation of astrocytes at the injury site repopulates the lesion with non-neuronal cells and secretes inhibitory proteoglycans, extracellular filaments and matrices that begin to form a glial scar around the lesion. This precedes a secondary phase of pathologies that radiate outward into healthy tissue causing diffuse demyelination, neuronal axon loss, excitotoxicity and neuronal cell death (Richardson, 2010, Namas, 2009, Lo, 2010). In both phases, the operative pathology involves the acute or delayed loss of mature neuronal networks and the upregulation of mechanisms which inhibit neo-neurogenesis and engraftment. However, in response to mechanical injury and ischemia, human and rodent brains themselves can up-regulate endogenous adult neural stem cell activity (Cayre, 2009, Lo, 2010). Neural stem cells (NSCs) which retain their multipotency into adulthood, reside in a few select regions of the brain and play a role in homeostatic neural tissue maintenance. NSCs provide a new source of neuronal and glial progenitor cells that can differentiate and replace lost neurons, astrocytes and oligodendrocytes (Jin, 2010). In rodents, neuroblast cells endogenously proliferate and migrate towards the anterior region of the brain to the olfactory bulb where they support the 3 high turnover rate for new interneurons. Likewise in animal models of stroke, neurodegenerative disease and mechanical injury, the major stem cell niches of the brain, such as the subventricular zone (SVZ), upregulates NSC proliferation and their egress out of the niche (Reekman, 2012, Cayre, 2009, Lo 2010, Zhang, 2011). This paramedic response to injury is maintained over the course of a lifetime, though notably this capacity diminishes significantly with age (Cayre, 2009, Chen, 2011). Previous studies have reported incidences of neural precursor cell migration from the cell niche over a year after insult, demonstrating the persistence of endogenous NSC recruitment (Cayre, 2009). Much of current translational neuroscience research focuses on characterizing and optimizing the transplantation of stem cells in acute and progressive neurotrauma animal models. This effort has produced many studies showing positive increases in functional recovery, making a strong case for hNSC transplantation as a future cellular treatment (Tsuji, 2010, Shimada, 2011, Lo, 2010, Reekman, 2012). Though the exact mechanism is yet to be determined, transplanted NSCs have been shown to survive in the host brain and migrate towards the site of injury. Engrafted cells demonstrated both molecular and electrophysiological markers of terminally differentiated cells (Tsuji, 2010, Shimada, 2011). Additionally, NSCs can modulate the T-cell activation response and secrete neurotrophic growth factors that support cell survival and neuroprotective functions (Lo, 2010, Reekman, 2012). Research in this field has also applied the technology behind growing NSCs from embryonic stem cell (ESC) lines to create a source of cells for research and potential clinical use (Reekman, 2012). Though the science is still far from supporting a standard sustainable supply of clinical grade NSCs, such a resource would eliminate the need for an allogeneic or autologous tissue harvest (Reekman, 2012). Several 4 animal trials have already reported successful integration of ESC derived cells following intracranial transplantation. (Denham, 2012, Tsuji, 2010, Kriks, 2011) Yet despite their regenerative capacity and trophic support, the endogenous recruitment of NSCs is still insufficient to significantly regenerate lost tissue and recuperate damaged neural functions. The inflammatory response and astrocyte hypertrophy creates an inhospitable microenvironment for recruited precursor cells, reducing their survivability and migration into the glial scar (Reekman, 2012, Cayre, 2009). These microenvironmental conditions that attenuate the recruitment of endogenous stem cells likely are also responsible for the limited engraftment and migration of exogenous stem cell transplants as well. Additionally, NSCs, such as those produced from the SVZ, are mostly confined to migrating within a designated structure or neural tracts, and thus their range of mobility is restricted barring a major disruption of its structural integrity. It is estimated that only 20% of migrating NSCs survive and, in a study of stroke model, only .02% of lost neurons were replaced by endogenous recruitment (Jin, 2007, Cayre, 2009). With respect to the clinical timeline post injury, the majority of the stem cell recruitment activity begins and peaks after injury onset but drops off shortly after (Lo, 2010). Beyond this refractory phase, the level of cumulative damage from secondary pathologies continues to increase over time such that beyond a certain threshold, intervention may no longer be effective (Lo, 2010). Efficient and robust migration of cells during this refractory period is therefore particularly important to mounting an effective intervention. Until there is an effective method for efficiently and quickly guiding cells to the site of injury, poor rates of cell engraftment and recruitment will remain an obstacle to functional NSC based regeneration. 5 One method to enhance NSC motility is stimulating with chemokines to direct migration. What was found early in the discovery of NSCs is their endogenous trophism towards glioma tumors (Cayre, 2009, Heese, 2005). Glioma cells release a myriad of growth factors and cytokines that promote cell proliferation such as basic fibroblast growth factor and epidermal growth factor (Heese, 2005, Chicoine and Silbergeld, 1997). However these were also found to induced a chemotactic response and for many mitogens, a corresponding chemokinetic effect as well (Heese, 2005, Chicoine and Silbergeld, 1997). In addition, extracellular ATP, which is a released in a paracrine fashion by neuronal and glial cells, has also been found to have a trophic and chemotactic effect on neural stem/progenitor cells (Grimm, 2010). These factors are also known to keep NSCs undifferentiated and to maintain multipotency, a state which is normally transient when outside of the stem cell niche. Because these chemokines NSCs, this study investigates the contributions of basic fibroblast growth factor, epidermal growth factor and extracellular adenosine triphosphate to increasing NSC motility. Basic fibroblast growth factor, or bFGF-2, is one of 20 members in the fibroblast growth factor (FGF) ligand family (Boilly, 2000). It is a standard component of embryonic and neural stem cell culture for maintaining pluripotency (Reekmans, 2011) but it was previously found to promote cell proliferation and have a chemotactic/chemokinetic effect on glioma cells and neural stem cells in vitro (Yamamoto, 1997, Heese, 2005, Erlandsson, 2004). Systemic infusion of bFGF and EGF after NSC transplantation shows greater migration towards lesion site demonstrating its effect on motility in vivo as well (Cayre, 2009). Endogenously, it is produced by neurons and astrocytes and is one of many neurotrophic factors releases following injury (Mattson and Scheff, 1994). Mechanistically, bFGF binds and activates its corresponding FGF receptor (Boilly, 2000) via phosphorylation from SH2 domain signal transducers. The receptor 6 activates the G-protein molecular switches Rac, RhoA and Rho kinase (which are critical to adhesion and lamellipodia formation during migration (Abe, 2007)) and enzyme kinases such as Src, Protein Kinase C and PI3-kinase (Boilly, 2000). Epidermal growth factor (EGF) is similar to bFGF in several ways. EGF is also a neurotrophic growth factor released from astrocytes in response to injury. It too has positive effects on cell proliferation and motility (Reekman, 2011, Grimm, 2010, Mattson and Scheff, 1994). EGF is a small peptide growth factor that binds to EGFR (a member of the receptor tyrosine kinase family of transmembrane receptors) (Boockvar, 2003, Liebmann2011). Similar to other RTK type receptors, it can activate downstream effector enzymes such as Stat3, Phospholipase C, the Akt pathway and the MAPK ERK pathway (Boockvar, 2003, Liebmann, 2011). Compared to the previous chemokines, extracellular adenosine triphosphate ATP has only been recently recognized as a cell signaling factor rather than as a metabolic product. The extracellular release of nucleotides, such as ATP and ADP, are mediated by most cell types as well as during cell death in a wound or stress scenario (Zimmerman, 2006, Siow, 2010, Burnstock, 2011). Nucleotide ligands are detected by two classes of purinergic receptors, P2X ionic channels and P2Y metabotrophic G-protein coupled receptors (Fischer, 2007, Ferrari, 2011). The family of receptors is composed of subsets of members with higher affinity to a specific nucleotide (i.e. ATP vs. ADP vs. UTP) (Zimmerman, 2006, Burnstock , 2011). Different classes of purinergic receptors trigger different signaling pathways regulate multiple cellular functions such as proliferation, migration, apoptosis, differentiation and self-renewal. Not surprisingly, specific subsets of purinergic receptor are found expressed only during the undifferentiated stage of neural stem cells (Siow, 2010, Burnstock, 2011). Extracellular 7 nucleotide-mediated purinergic signaling plays a role in regulating the migration of progenitor and stem cells (Cayre, 2009, Ferrari, 2011, Zimmerman, 2006). Specifically, ATP and ADP activate several P2Y receptors that trigger intracellular pathways often associated with migration of neural cells (Liu, 2008, Agrestia, 2005). These include the phosphorylation of Phospholipase C, MAPK ERK signaling pathways, focal adhesion kinase and induction of temporary intracellular Ca2+ spikes (Burnstock, 2010, Zimmerman, 2006, Grimm 2010). A recent study of hMSCs also shows purinergic signaling 1) upregulates genes involved in cytoskeleton reorganization while downregulating genes in cell adhesion and proliferation, and 2) significantly increases spontaneous migratory behavior even in the absence of chemokines (Ferrari, 2011). Extracellular ATP-activation can produce transitory Ca2+ influxes and promote the migration of oligodendrocyte progenitor and differentiated cells (Siow, 2010, Burnstock, 2011, Cayre, 2009). In addition to chemical cues, physical stimuli, specifically, electric fields (EFs), can also direct cell division and migration direction, in some cases as an ‘overriding’ dominant cue (Zhao, 2006, 2009, Forrester, 2006). This is interesting particularly because by nature and function, the brain itself is an electric organ and NSCs theoretically exist within an environment of endogenous electrical activity. To explain wound directed electrotaxis, an epithelial wound models best illustrates a simplified scenario of wound healing in endogenous EF (Zhao, 2009, Forrester, 2007, Figure1). The voltage potential behind electric fields originates from the ion channels and transporters on the apical and basal sides of the membrane, mediating the transmembrane ion concentration (Na+, K+, and Cl-). The apical side of the tissue typically maintains a net opposite charge relative to the basolateral side, creating a transepithelial voltage 8 potential. This voltage potential can be measured in skin, corneal epithelium, respiratory, gastrointestinal, urinary and retinal epithelial tissues as well (Zhao, 2009, Forrester, 2007). Disruption of the cell layer (i.e. a wound) presents a breach in this charge separation, allowing ions to migrate and circumvent this barrier creating an electrical current and the wound EF. Endogenous wound EFs have been recorded in multiple systems including, human and rodent skin and corneal wounds (Zhao, 2009). Its strength depends on wound size and follows a voltage gradient from as high as 140mV along the wound edge down to 0mV towards the center of the wound as measured in guinea pigs and human skin (Zhao, 2009, Forrester, 2007). 9 Transport of Ions Na + Transepithelial Potential 0mV ++ Wounding -- - Transport of Ions Na+ Cl - +25~40m V Wound Electric Fields Transe - -- + + 2 5uA/cm Figure 1 Mechanism generating electric field at wound site. Epithelial model of membrane potential shows segregation of positive and negatively charged ions by a membrane barrier creating a voltage potential. Breach in the barrier (i.e. wound) creates an electrical current in which cells may migrate (as illustrate by bold outlined cells). Cells shown here migrate towards the cathode but different cell types migrate to different poles. (Image created based on figure by Zhao (2009) Cl- +25~ 10 Near the wound edge, cells in mitosis detect and divide along the EF vector and migrate along available surfaces in the direction of the EF vector from the more positive ‘pole’ towards the wound edge with an oppositely charged ‘pole’ (Zhao, 2009, Forrester, 2007). As the wound remains open, the wound EF is maintained by intact peripheral and basolateral cells surrounding the wound that preserve homeostatic ionic concentrations. This keeps the wound edge differentially charged compared to the void of cells and voltage at the wound center until the epithelial barrier is restored (Zhao, 2009, Forrester, 2007). There are several pivotal studies that also recapitulate electrotaxis in vitro using exogenous EFs. The overriding effect of an EF in vitro was most dramatically demonstrated in a scratch wound assay where cells in unison can switch migration direction when an EF was applied and then reversed. Collective electrotaxis occurred even away from the wound against the direction of contact inhibition pressures and chemical gradients (Zhao, 2009). Electrotaxis was also confirmed not to be a chemotaxis artifact when media cross flow experiments demonstrated that in spite of consistent fluid flow to remove any confounding chemotactic gradients created via electrophoresis, cells maintained a directed migration response towards one electrical pole in the presence of an electrical stimulus (Song, 2007, Yao, 2011). In vitro, cells in electrotaxis not only migrate directionally but in some cases, faster (Meng, 2010, Zhao, 2009). This electrotactic behavior has been observed in several different cells types (including keratinocytes, corneal and vascular endothelium and neurons) and in more recent studies with stem cells and early progenitor cells as well (Arocena, 2010, Zhao, 2009, Meng, 2010, Yao, 2011, Zhang, 2011). Electrotaxis in an exogenous DC electric field has been observed in cultured cell monolayer models, stratified tissue and cultured 3D environments (Zhao, 2009, Zhang, 2011). Surprisingly, endogenous processes such as directional nerve sprouting, vessel 11 growth and neurogenesis respond to this force as well (Song, 2004, 2007, Zhao, 2009) and possibly even prenatal development (Yao, 2011, Saunders, 2005). To date, it is not know how cells detect EFs or if there is an EF ‘receptor’ however several intracellular pathways regulating electrotaxis have been identified. Cells exhibit polarized intracellular gradients of actin fibers and adhesion molecules towards the leading edge (Zhao, 2009). Similar to chemotaxis signaling pathways, electrotaxis (of neutrophils, keratinocytes, NPCs and epithelial cells) shows polarized intracellular gradients of activated MAPK signaling, Src, Pten and PI3K towards the leading edge in parallel to the EF vector (Meng, 2010, Zhao, 2002, 2009). Interestingly, cells in EF also demonstrate an asymmetrical concentration of membrane lipids, surface receptors, and polarization of Golgi apparatus suggesting a biased presentation of decorated cell membrane and ligand detection at the leading edge (Meng, 2010, Zhao, 2009). Exogenous EFs have also been known not only to alter distribution but to modulate the expression of signal receptors such as EGF receptors (Zhao, 2002). Additionally, NPCs and neural cells display increased intracellular Ca2+ signaling as mediated through voltage-gated Ca2+ channels and endoplasmic reticulum stores (Yao, 2011, Meng, 2010). All of these intracellular signals are represented during chemotaxis and migration, verifying not only the molecular mechanisms behind electrotaxis but also the potential signal crosstalk between electrical and chemical migration cues. It suggests that there may be an additive benefit in improving directional migration due to convergent intracellular signaling and transactivation of neighboring receptors. During wound healing, it is likely that both neurotrophic chemical release and wound electric field are present. NSCs migrating in this signaling milieu will likely be exposed to both stimuli and could possibly be an endogenous component of their recruitment to the injury site. A few possible convergent transduction pathways include MAP kinase ERK pathway, Src, PI3K 12 signaling pathways and an increase in intracellular Ca2+ waves (Cayre, 2009, Zhao, 2002, 2009, Meng, 2010). It therefore begs the question if exogenous chemical and electrical co-stimulation would improve NSC migration efficiency. Though research in this area is very limited, it suggests an additive chemokinetic effect may be co-opted to improve the migration and engraftment level of NSC transplants, thereby enhancing their regenerative and neuroprotective functions. In addition, as a utility within the toolkit of stem cell-based biotechnology, the ability to direct stem cell migration has considerable possibilities in future biomedical and basic research applications. In this study, we investigated migratory behavior of hNSCs in vitro in response to exogenous chemical (bFGF, EGF and ATP) and electrical stimulation. Our studies suggest that direct current electrical field significantly increases cell motility and directionality to a greater degree than chemical stimulation alone. However using a co-stimulation modality enhances directional migration but not due to additive motility. 13 MATERIALS & METHODS Culturing H9-ESC Derived NSC for Migration Analysis Human embryonic stem cell line (H9) derived neural stem cells (NSCs) were kindly provided by Dr. Jing Liu (Institute of Regenerative Cures). Liquid N2 frozen cells are rapidly thawed at 37°C before immediately resuspended in warm media to dilute the cytotoxic cryoprotectant, dimethyl sulfoxide (DMSO). Cells are collected by pelleting via centrifugal force and resuspended again with warm DMSO-free media. Multicellular clusters that shield ‘core’ cells from growth factor signaling are likely to differentiate and produce impure cell populations. Therefore a completely dissociated cell suspension is important to multipotent stem cell culture by allowing uniform growth factor signaling necessary to maintain their phenotype and inhibit the propensity to differentiate. Cells are seeded at a high density to promote paracrine signaling inducing cell survival and proliferation. NSCs are seeded on .003% poly-L-Ornithine (Sigma) and 10ug/ml laminin (Sigma) coated polystyrene tissue culture plates (BD Falcon) and cultured at 37°C, 5% CO2, 100% humidity in Neurobasal Medium (Gibco) supplemented with 100uM MEM nonessential amino acids (Invitrogen), 2mM L-glutamine (Invitrogen), 1% N2 Supplement (Invitrogen), 1% penicillin/streptomycin (Invitrogen), .1% B27 Supplement (Gibco), 20ng/ml basic fibroblast growth factor, bFGF (Invitrogen), and 10ng/ml epidermal growth factor, EGF (R & D Systems). Media is changed every two days to remove waste products and replenish nutrients and growth factors, allowing NSC cultures to expand four to fivefold into confluent cell monolayers within 5-6 days. A confluent culture can then be passaged and divided into multiple cultures to increase cell stocks. During passage, intercellular connections are digested with a 14 gentle proteolytic, collagenolytic solution, Accutase (Stem Cell Technologies). Cells are allowed to recover for a minimum of two days post passage to restore surface receptor, remove dying cells and allow entry into an active proliferation phase. Cells are used between the 4th and 10th passage to prevent any major changes in morphology or migration that may occur due to cumulative cell or DNA damage that may occur during passaging. At least 12hrs prior to experimentation, NSC are starved in reduced growth factor culture media (5ng/ml bFGF and 2.5ng/ml EGF) to magnify effects of chemical agonists that may share convergent signaling pathways with culture media growth factors. Chemical stimulation of NSC migration and motility 3 x 104 cells are plated in laminin coated 24 well plates (Corning Costar) with 300-500ul of 25% reduced growth factor media to adhere in 37°C, 5% CO2, 100% humidity culture conditions. After one hour cell seeding period, cells are adapted to 37°C atmospheric conditions and CO2 independent media (Gibco) supplemented with 1% Penicillin/Streptomycin (Invitrogen), 2mM L-glutamine (Invitrogen), here after referred to as ‘CO2 Media’. This media change is necessary because exposure to atmospheric conditions alkalizes culture media beyond a tolerable range for NSCs, therefore cells must be maintained in a buffering media that maintains stable neutral pH for multiple hours. After CO2 adaptation, cells are observed for two hours before carefully replacing media with 500ul CO2 media containing the chemical agonist. Chemical stimulants used in the study include adenosine triphosphate disodium salt (Sigma) diluted in ddH20, 20ng/ml bFGF (Invitrogen) in .1% bovine serum albumin, BSA (Sigma), and 10ng/ml EGF (R & D Systems) in 10mM acetic acid. Cell migration was recording for at least two hours following agonist addition. 15 Electric field (EF) stimulation of NSC migration and motility a) Chamber Preparation Electrotaxis chamber preparation is detailed previously (Zhao et al., 2010, Song et al.2007). [Illustrated in Figure 2 below] Briefly, two 10 x 22 mm chamber regions coated with laminin, are flanked by 8 x 22mm No. 1.5 coverslips (VWR) secured by high vacuum grease (Dow Corning). Media reservoirs are created with vacuum grease borders at the head and tail openings of the chambers to control media flow. Following starvation period, NSCs are harvested with Accutase and 3 x 10^4 cells in 200 to 300ul media is evenly seeded between cover slips. Following cell adhesion to surface, media is changed to CO2 media. A 22 x 30 mm sterile coverslip roof is secured over both cover slips with grease to seal the EF chamber, leaving a boxlike tunnel of uniform dimensions between reservoirs for media flow-thru. Reservoirs are filled with approximately 3 ml CO2 Media and NSCs in EF chambers allowed to adapt to atmospheric conditions at 37°C for one hour before time lapse recording of self-control treatment. 16 ~.1mm thick Figure 2 Construction of EF Chamber. Schematics of dish containing two electrotaxis chambers based on design created by Song et. al (2007) that can be simultaneously imaged during the same time period. Image on left shows the construction and dimensions of dual electric field (EF) chamber with cells seeded in the chamber center. Media reservoirs on either side additional media to buffer small temperature and pH changes in experiment and act as a conduit for direct current to be applied across cells. Image on right shows magnified image of an EF chamber and approximate placement of the agar bridges. 17 b) Application of Direct current EF Stimulation to hNSCs EF application protocol is described in greater detail by the cited literature in references (Zhao, Song et. al, 2007) [See Figure 3 for illustration of this system]. Briefly, direct current (DC) power source (Consort EV265) is connected via leads to Ag/AgCl electrodes submerged in 1X Steinberg’s solution. 1 to 1.5% Agar-Steinberg filled glass bridges contact Steinberg solution which contains ionic salts to conduct current, and CO2 media reservoirs forming a completed electrical circuit and EF vector through the seeded cells. Additionally EF voltage is significantly reduced and dramatic changes in pH are buffered. Prescribed voltage strengths across chamber are applied and monitored during recording. Two sets of agar bridges and Steinberg’s solution beakers are required for experiments applying EF to both EF chambers simultaneously. 18 Figure 3 Electrotaxis Experiment Set Up. Stem cells seeded in the electrotaxis chamber are exposed to DC current via agar bridges immersed in Steinberg solution. Current is conducted from power source through silver electrodes and Steinberg solution for a complete circuit. The double chamber design allows for simultaneous imaging of a no EF control group. 19 Time lapse Recording of NSC Migration Behavior Cell migration is imaged with Zeiss Axiovert 40CFL inverted microscope and motorized stage (BioPoint 2 Ludl Electronic Products) in a 37°C temperature controlled enclosure. Time-lapsed images are recorded with Hamamatsu digital camera every 10mins and images are compiled into videos using Simple PCI 5.0 imaging software (Hamamatsu). Quantification of NSC Migration Parameters with Stimulation Modalities Compiled image stacks are corrected for background tremble with Metamorph 7.0 imaging suite (Molecular Devices) in order to minimize any augmentation of data due to shaking of the motorized stage. Migration speed and distance for 50 to100 cells per treatment are calculated by manual tracking of migrating cells with ImageJ analysis software (NIH, Wayne Rasband). Quantification of migration parameters was performed with ImageJ 1.46 and Microsoft Office Excel 2007. Track speed is treated as a ‘spontaneous’ speed, of a ratio of total distance travelled in time interval divided by time interval. Migratory distance is measured as accumulated distance (total distance travelled) and Euclidean distance (net geometric displacement from the start site). Directedness of migration is measured as cosine of the angle between the migration path vs. electric field vector. Statistical Analysis of Migration Parameters Data is compiled using Microsoft Office Excel 2007 and statistical significance was determined with IBM SPSS Statistics 19 by with-in subject repeated measures ANOVAs and Bonferroni confidence interval correction or paired and unpaired Student’s T-test with and significance for both set at p< .05. 20 RESULTS H9 hESC derived NSCs display characteristic designation and markers of NSCs The current protocol for deriving NSCs from ESCs requires weeks of careful manipulation of the extracellular signaling environment. Embryonic stem cells colonies are detached from adherent culture and maintained in floating suspension to induce the formation of an embyroid body, a sphere comprised of cells differentiating into the three primary germinal cell layers. Reattachment of an embryoid body to an adherent culture surface signals cells to further differentiate and recapitulate the process of neural tube development as seen by the formation of a neural rosette. Columnar cells at the rosette center express the NSC markers, nestin (an intermediate filament) and sex determining region Y - box2 (SOX-2) (a pluripotency associated transcription factor). When extracted and cultured, H9-NSCs demonstrate the characteristic bi/tripolar cell morphology (Figure4a) and demonstrate the capacity to form 3D neurosphere-like aggregates (data not shown). In addition, they display cytoplasmic staining for nestin and nuclear staining for SOX-2 (Fig 4b/c/d, photos provided by Dr. Jing Liu). 21 a) 10x Bright field Phase contrast c) Nestin b ) Merged d ) SOX2 Figure 4 H9 NSC exhibit motile phenotype in absence of stimulation. a) Cultured H9 hNSCs starved to 25% of culture growth factors display bipolar and tripolar cell morphology characteristic of neural stem cells. b) NSCs are cultured and passaged til higher passages numbers (12th) before staining for nestin and SOX-2 expression. Merged labelling shows localization of nestin and SOX-2 staining in relation to cell. c) Nestin specific staining coincides with cytoplasmic region of cells. d) SOX-2 staining is localized in nuclear compartment and is round coinciding with shape of nucleus. 22 hNSCs display motile but non-linear, highly tortuous random migration behavior In vitro migration studies demonstrate fundamental migratory behaviors unique to specific cell types. To observe basal migration behavior of hNSCs, cells were plated on a laminin coated surface and maintained in a reduced growth factor, serum-free CO2 media as they are observed in a timelapse digital video recording. The basal level of migration was measured as a function of speed and distance travelled. In the absence of exogenous stimulation, NSCs are highly motile (Figure 5). However the extent of cell motility is heterogeneous between cell passages and even between individual cells within a population (Fig 6a). However when averaging a significantly high cell count, a defined migration trend and a small standard error can be achieved. Cells can also migrate quite quickly, at its peak travelling 50.88 um/hr (+1.82 um/hr) but slowing as time progresses (at 8 hrs 40.188um/hr + 1.77, p=.00015, d.f=172) (Figure 6b). However track speed is a dynamic parameter, speed measurements changes significantly when measured on an hourly basis, appearing even more stochastic so at shorter time points with alternating higher and lower speeds. In order to establish a stable baseline in which to compare treatments but still show overall trends, ‘spontaneous’ migration speeds are measured in 2hr intervals. Cells also travelled a significant distance, nearly 4/10th of a millimeter (369.95um + 11.62) cumulatively over an eight hour period (Figure 5, 6c). However the actual distance travelled away from the cell’s starting site is dramatically less (89.64um + 4.59) (Figure 6c), demonstrating the migratory path is non-linear or tortuous and as seen in time lapsed recording, sometimes circling around it’s start point (Figure 5). Additionally the directionality of cell migration is low (.019 + .053) as expected due to the cell’s random migration in the absence of a directional cue (Fig 5, 6d). 23 No Stimulation-2hrs No Stimulation-6hrs No Stimulation-4hrs No Stimulation-8hrs No Stimulation-6hrs Circling tortuous migration pattern Figure 5 H9 NSCs display an actively motile but non-linear random migration in a heterogeneous population. Time-lapsed analysis of manually tracked cell migration over 2hr intervals displays distance and tortuosity of migration paths. No Stimulation-8hrs 24 a) b) c) d) Figure 6 hNSCs speed stable for several hours but inefficient due to tortuous random behavior. a) Cells migrating in absence of exogenous stimulation migrating for 8 hours. Wide distribution of migration speeds of cells within a sample population but little change over time. b) Average track speed of population peaking at 50.88um/hr (+ 1.82um/hr) not significantly different for first six hours but decreasing after eighth hour (40.18um/hr + 1.77um/hr, df=2.290, F=14.519, p=1.878 x 10-7). c) Cells migrate cumulatively after 8hrs on average 369.96 um (+ 11.62 um, df=3, f=14.519, p=4.32 x 10-9) while net Total Euclidean displacement of cells after 8hrs is 89.64 um (+ 4.59 um) migrating a significant distance away from starting point (df = 2.208, F=90.414, p=2.513 x 10-35). d) Directedness of migration paths is measured from 1 (completely parallel with horizontal axis going from left to right) to 0 (completely parallel with vertical axis). Cells travelled in absence of stimulation with directedness value of 0.019 (+ 0.053), due to randomness of migration, a wide range of sample values results in a small average and larger standard error. Directedness did not significantly change over time (df = 2.23, F= 2.006, p=0.130.) 25 Chemical stimulation alone shows no significant change in cell motility NSCs were starved of growth factors, to reduce the level of growth factor signaling associated with culture conditions that may interfere with their effect in migration and sensitize cells. In order to preserve NSC multipotency, growth factor starvation was shortened to 12 hours rather than 24 hours typically found in other migration studies. In vitro chemotaxis studies typically demonstrate an effect by looking for an increased occurrence of NSCs in the region of the highest ligand concentration. However the majority of in vitro studies employ a transwell experimental system thereby creating an artificial concentration gradient to study agonist guided chemotaxis. To study the chemokinetic effect of bFGF, EGF and ATP, a uniform concentration is presented following a two hour basal self-control migration period. When exposed to concentrations previously reported to direct cell migration (Heese, 2005, Grimm, 2010), bFGF (at 5ng/ml and 20ng/ml), EGF (at 1ng/ml, 5ng/ml and 20ng/ml) and ATP (at 1uM, 10uM, 100uM), hNSCs appear to have no significant positive increases in track speed (Figure 7 and 8). In some cases, the track speed slightly decreases as with bFGF (Figure 7a) and 1 and 10uM ATP (Figure 7b, Table 1). The self-control migration period for EGF stimulation could not be recovered and is therefore not included however a larger p-value was noted compared to the vehicle suggesting a more significant change in speed compared to the control (Figure 8, Table 1c). However in either case, the small degree of change may have little to no biological significance with regards to motility. 26 b) a) Add Agonist Agonist d) c) b) * * Add Agonist Figure 7 Chemical stimulation of growth factor-starved hNSCs with bFGF and extracellular ATP shows no increase in chemokinetic migration. Specifically a & c) hNSC migration distance and b & d) speed during a 2hr self-control period before changing to CO2 media with bFGF vehicle (.1% bovine serum solution), 5ng/ml and 20ng/ml bFGF or ATP vehicle (double distilled water), 1uM, 10uM and 100uM ATP. Track speed slows significantly in the 4nd hour for 5ng/ml and 20ng/m bFGF (Table 1a). 27 Figure 8 Chemical stimulation of growth factor-starved hNSCs shows no increase in chemokinetic migration in EGF stimulation. hNSC migration distance and speed for recorded for a 2hr self-control period before changing to CO2 media with EGF vehicle (10mM acetic acid solution), 1ng/ml, 5ng/ml and 20ng/ml EGF. No self control data is available to show. Therefore comparisons between the 2hr and 4hr timepoints were made and against values from the vehicle treatment 28 Table 1 Values of significance between NSC track speeds with chemical stimulation a) P-values from repeat measure ANOVA test of cell motility following exposure to bFGF vehicle (.1% bovine serum solution), 5ng/ml and 20ng/ml bFGF b) ATP vehicle (double distilled water), 1uM, 10uM and 100uM ATP, c) EGF vehicle (10mM acetic acid solution), 1ng/ml, 5ng/ml and 20ng/ml EGF via repeated measures ANOVA with Bonferroni confidence correction a) Group Period N df F bFGF vehicle 5ng/ml bFGF 5ng/ml bFGF 20ng/ml bFGF 20ng/ml bFGF SC vs. 4hr SC vs. 4hr 143 192 192 230 230 2 1.711 1.846 - 2.472 4.494 8.375 - Group Period N df F ATP Vehicle ATP Vehicle ATP Vehicle ATP Vehicle 1uM ATP 1uM ATP 1uM ATP 1uM ATP 10uM ATP 10uM ATP 10uM ATP 10uM ATP 100uM ATP SC vs. 2hr SC vs. 4hr 2hr vs. 4hr SC vs. 2hr SC vs. 4hr 2hr vs. 4hr SC vs. 2hr SC vs. 4hr 2hr vs. 4hr - 154 154 154 154 164 164 164 164 169 169 169 169 170 1.645 1.734 1.799 2 4.068 9.375 4.708 2.185 Group Period N df F EGF Vehicle 1ng/ml EGF 5ng/ml EGF 20ng/ml EGF - 246 229 306 229 1 1 1 1 26.22 16.48 60.36 61.23 Sig (p) values 0.039 8.65 x 10-4 Sig. (p) with-in subject effects 0.0862 0.016 4.068 x 10-4 - Sig (p) values 0.058 0.093 1 1.08 x 10-4 0.009 1 0.004 0.239 0.837 - Sig. (p) with-in subject effects 0.025 2.526 x 10-4 0.012 0.114 Sig (p) values - Sig. (p) with-in subject effects 6.143 x 10-7 6.74 x 10-5 1.21 x 10-13 1.88 x 10-13 b) c) 29 Electrical stimulation significantly increases hNSC motility and directionality of migration Despite cell-to-cell differences in migration behavior, most cells exposed to electric field responded collectively (Fig 9a). On average, hNSCs in EF migrated a longer accumulated distance compared to the no EF growth factor starved control (Figure 9b, Table 2) and a greater net Euclidean distance from it’s starting point (Figure 9c). Comparing the two EF strength conditions, though their accumulated distances are similar, cells in 300mV/mm migrates a longer Euclidean distance compared to100mV/mm EF. This suggests the migration paths of cells in a lower voltage strength involves more circling or non-linear migration behavior. The track speed is also significantly higher compared to the no EF control (Figure 9d, Table 2). Most strikingly, the migration of most cells is uniformally directed towards the negitive cathodal pole (Figure 10). Compared to the control treatment which shows cell disperal radiating outward in all directions, cells in EF shows migration paths significantly skewed along a horizontal axis (Figure 10). Thus the directedness value of cells in EF is also significantly higher compared to no EF and directedness is corresponds to voltage strength (Figure 11a, Table 2). This is also reflected in the net x-axis displacement of the migration tracks of cells in EF (Figure 11b). However, it was also observed in the EF stimulation groups, compared to the control group showed the CO2 media alkalized after recording overnight, that the CO2 media had alkalized sooner and was acclerated with a higher voltage strength (data not shown). This pH change required a media change in the EF chamber, which was also conducted for the control group, and an adjustment of the EF field approximately every two hours. A delay in this change usually resulted in mass cell death (data not shown). 30 b) a) No Electric Field – 4hrs Apply EF c) Apply EF 300mV/mm Electric Field – 4hrs + _ d) * * Apply EF Figure 9 NSC motility is enhanced when exposed to electrical field. a) Baseline hNSC migration is observed in self-control for two hours before applying 100, 300mV/mm or no EF for four hours EF stimulation. Linear migration to cathode is seen. b) Quantification of total accumulated migration distance of average cell with no EF, 100mV/mm and 300mV/mm EF. c) Quantification of net Euclidean distance before and during four hours of EF stimulation shows more linear migration path compared to no EF or a lower voltage. d) Quantification of ‘spontaneous’ track speed during EF stimulation. Motility dramatically increases with application of EF. 31 a) Self-Control 2hr EF 4hr EF No Electric Field b) Self-Control 300mV/mm EF 2hr EF + 4hr EF - + - Figure 10 hNSC migration is less tortuous and more directed towards cathode when exposed to electrical field. a) Cells in the absence of EFs show no change even distribution of cells migrating outward in all directions. b) Conversely, cells in electrical field travel predominantly travel towards the right in the direction of the cathodal pole. Plot graphs of cell migration show migration path of each treatment which quantify the number of cells with net displacement to the left (in black, towards the anode) or the right (in red, towards the cathode). Cross represents center of mass for all cell migration 32 a) Apply EF b) Apply EF Figure 11 hNSC migration directedness is quantified showing greater directional migration towards cathode when exposed to electrical field. a) Quantification of directedness of cell migration with no EF, 100mV/mm, 300mV/mm which is most directional at 300mV/mm EF. b) Net migration distance of hNSCs travelled along x-axis direction with and without EF stimulation. 33 Table 2 Values of significance between NSC speeds and directedness with electrical stimulation a) comparing ‘spontaneous’ track speed during no EF stimulation, 100mV/mm and 300mV/mm EF stimulation and b) quantification of directedness of cell migration with no EF, 100mV/mm, 300mV/mm via repeated measures ANOVA with Bonferroni confidence correction. a) Group Period N df F Sig. (p) No EF Ctrl 100mV EF 100mV EF 100mV EF 100mV EF 300mV EF 300mV EF 300mV EF 300mV EF SC vs. 2hr SC vs. 4hr 2hr vs. 4hr SC vs. 2hr SC vs. 4hr 2hr vs. 4hr 218 128 128 128 128 190 190 190 190 1.92 2 1.42 59.17 Group Period N df F Sig. (p) No EF Ctrl 100mV EF 100mV EF 100mV EF 100mV EF 300mV EF 300mV EF 300mV EF 300mV EF SC vs. 2hr SC vs. 4hr 2hr vs. 4hr SC vs. 2hr SC vs. 4hr 2hr vs. 4hr 202 128 128 128 128 190 190 190 190 1.92 1.35 2 - 1.42 35.49 93.11 - 1.05 x 10-6 1.021x 10-9 0.001 8.09 x 10-13 7.40 x 10-16 4.38 x 10-4 Sig. (p) with-in subject effects 0.24 7.99 x 10-22 1.11 x 10-18 2.53 x 10-10 0.0019 1.80 1.42 x 10-30 93.11 -25 7.20 x 10 5.56 x 10-20 1 b) Sig. (p) with-in subject effects 0.242 1.65 x 10-10 1.16 x 10-16 - 34 Chemical and electrical co-stimulation increases hNSC directedness but not motility consistently In the wound healing microenvironment, both chemical and electrical signaling occur simultaneously and in all likelihood, act synergistically with each other. To investigate the effect of this co-stimulation, we applied a 200mV/mm EF to cells in media containing 20ng/ml EGF. 200mV/mm EF strength was applied to attenuate some of the physical changes to the media as a result of a higher voltage. Due to the limited availability of data and large deviation in values between experiments, all results are presented to demonstrate the level of variation between these experiments but also to highlight the trend seen in both. The basal cell migration speed for this set of experiments is near or higher than peak migration speeds seen in the control assay (Figure 12a). So from the beginning, the motility of these cells are higher than previous experiment’s cultures. However because the self-control migration speeds between the EF stimulation only vs. chemical and EF combined stimulation condition were similar, the trend they exhibit appears valid (Table 3). With the application of 200mV/mm EF, the migration speed jumps significantly for during a two hour period but eventually decreases back to near baseline (Figure 12a). Similar to the EF stimulation experiments, the directedness of migration also increases significantly and the migration is more linear along the x-axis towards the negative cathodal pole compared to its self control baseline (Figure 12b, 13, Table 3). Compared to the chemical-electrical co-stimulation modality, directedness is increased and the migration path of most cells becomes more linear after the application of EF (Figure 12b, 13). However there is no overall significant increase in the accumulated distance travelled. 35 a) Apply EF Apply EF Trial 1 Trial 2 b) Apply EF Apply EF Trial 1 Trial 2 Figure 12 EGF combined with EF stimulation increases migration directionality but does not consistently increase motility. a) Track speed of NSCs during EF only or EF with 20ng/ml EGF stimulation found to migrate at similar speeds. b) Migration Directedness of migration in 200mV EF vs. 200mVEF +20ng/ml agonist. Directedness in the co-stimulation modality is higher compared to EF alone. 36 Apply EF Trial 1 Apply EF Trial 2 Figure 13 Trends shows greater net horizontal displacement in EGF combined with EF stimulation. Greater cathodal migration compared to just EF alone. 37 Table 3 Values of significance between NSC speed, directedness and distance when comparing electrical stimulation with chemical-electrical co-stimulation. P-values of significance of a) track speed of NSCs during EF only or EF with 20ng/ml EGF stimulation, b) migration directedness of migration in 200mV EF vs. 200mV/mm EF +20ng/ml agonist in co-stimulation modality compared to EF only. P-values are derived via paired and unpaired Student’s T-test. a) Trial Group Period N 1 1 1 1 1 1 2 2 2 2 2 2 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr 87 87 87 57 57 57 80 80 80 77 77 77 Trial Group Period N 1 1 1 1 1 1 2 2 2 2 2 2 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr 87 87 87 57 57 57 80 80 80 77 77 77 Sig. (p) with Self Ctrl 1.38 x 10-8 .991 4.85 x 10-9 .044 .237 2.44 x 10-6 .022 .407 Sig. (p) between subject - EF only .774 .0629 .0923 .826 .051 .0209 Sig. (p) with Self Ctrl 3.65 x 10-15 2.371x10-15 2.16 x 10 -10 2.8 x 10-11 5.46 x 10-6 4.36 x 10-5 1.99 x 10-9 1.76 x 10-8 Sig. (p) between subject - EF only .57 .15 .021 .509 .0035 .0047 b) 38 Table 4 Values of significance comparing net displacement of EF only to chemical-electrical costimulation. P-values of significance compares Trials 1 and 2, 200mV EF vs. 20ng/ml EGF and 200mV/mm EF, derived via paired and unpaired Student’s T-test. Trial Group Period N 1 1 1 1 1 1 2 2 2 2 2 2 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 200mV/mm EF only 200mV/mm EF only 200mV/mm EF only 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF 20ng/ml + 200mV/mm EF SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr SC SC vs. 2hr SC vs. 4hr 87 87 87 57 57 57 80 80 80 77 77 77 Sig. (p) with Self Ctrl 2.20 x 10-18 3.24 x 10-18 2.10 x 10-15 1.02 x 10-15 2.32 x 10-9 1.02 x 10-7 2.6 x 10-18 3.8 x 10-16 Sig. (p) between subject - EF only .245 .031 .002 .36 .0002 .0001 39 DISCUSSION Where endogenous tissue repair efforts fail to induce a functional recovery following neurotrauma, NSC transplantation shows potential for better functional recovery and tissue repair. Adult neural stem cells are widely thought to serve as a reservoir for new neural cell generation with an ambulatory function during neurotrauma. However a major obstacle in stem cell transplants is the efficient migration of cells to the site of injury to allow engraftment at injury site. Though many studies exploit chemotactic gradients or methods of sensitizing chemokine receptors to direct cell migration, fewer have looked at the use of physical cues to direct cell migration. Even fewer have applied an electric and chemokinetic co-stimulation condition. The relevance of such a study is that though electrical fields alone have been shown to direct cell migration, in reality, it is unlikely that chemical or electrical migration cues act independently in an in vivo wound healing setting. Our studies examined how NSC migration behavior is affected by chemical, electric and combined stimulation modalities. Without any exogenous stimulation, our observations confirmed the intrinsic motility of hNSCs. Our in vitro migration model is a biologically minimal environment because it does not provide an optimal network of adhesion substrates, surfaces, interstitial fluid flow, 3D intercellular cues or microenvironmental signaling found in an in vivo environment. Yet NSCs can migrate substantially, nearly 4/10th of a millimeter in just six hours on a permissive substrate, laminin, which is commonly associated with blood vessels and astrocytes. Additionally these cells were previously starved of bFGF and EGF growth factor signaling suggesting that few signaling factors are required in order to see significant NSC migration. There are also a limited number of studies that measure time-lapsed migration parameters such as distance and speed. 40 Compared to published rodent in vivo measurements, the hNSC migration speed found here is similar to those in vivo which fluctuates between 20-70um/hr and displays a similar stochastic migration behavior of alternating fast and slow migration periods (Bovetti, 2007). It suggests that our in vitro migration model allows for similar migration patterns as those seen in vivo. Our experiments suggest that growth factor starved NSCs when exposed to a chemical motogen in the absence of a concentration gradient, exhibits no significant positive chemokinetic change in baseline migration parameters such as migration speed or distance. In actuality, EGF signaling appeared to slow cell motility slightly. The chemical agonists used in this study are known to guide cell migration via chemotactic gradients (Heese, 2005, Zimmerman, 2006). However to study a chemokinetic effect, no concentration gradient was used and instead, a uniform ligand concentration was applied and expected to trigger greater, though random, cell motility. Additionally because these cytokines signal convergent intracellular signaling pathways such as the ERK and MAPK pathways also triggered during electrotaxis, a positive migration change was expected. Within the context of these studies, our negative results could point to several important ideas: Firstly, growth factor starvation beforehand may have diverted the cellular program from migration towards proliferation. Pre-experimental starvation is used to distinguish the lingering effects of cell culture growth factor signaling from the change in motility during experimentation. However, the cytokines we tested for chemokinetic properties are also involved in maintaining cell survival and proliferation as it is the case that most mitogens also have an effect on motility. They even require activation of similar intracellular factors such as protein kinase C (PKC) and ERK2 in bFGF (Boilly, 2000), EGF and ATP signaling. For pleiotrophic factors such as bFGF, the status of cell health and cell number can direct the receptor signal 41 towards regulatory pathways associated with proliferation (Boilly, 2000). It is possible that a pre-starvation period may change hNSC in vitro migration to a ‘nutrient seeking’ motile behavior. But upon exposure to neurotropic levels of environmental mitogens (such as a media change containing a high concentration of agonists), the cells may slow or stop migrating. Thus the activation of the same surface receptor may signal more cells to stop and divide rather than migrate, lowering the average migration speed for the sample population. Secondly, activation of ligand receptors is very tightly regulated. Upon ligand binding and activation of surface receptors, they are quickly endocytosed and degraded or uncoupled from their G-protein signaling intermediate (Bennett, 2011). The presence of a continuous uniform chemical signal may induce extended receptor desensitization; ‘communicating’ to the cell it has arrived in a region sufficient to support survival. This is one regulatory mechanism that prevents continuous stimulation by preventing excess presentation of active surface receptors on the cell membrane (Bennett. 2011). Likewise, there may be extracellular methods of regulation that prevent excessive signaling by degrading ligands in the extracellular space as is the case for extracellular ATP and ectonucleotidases (Zimmerman, 2006). Conversely, when looking at cellular mechanisms behind cancer metastasis, more than 40 oncogenes encoding tyrosine kinase surface receptors for chemokines such as bFGF, EGF and hepatocyte growth factor (HGF) are found to be overexpressed (Mareel & Leroy, 2003). This would appear to be an effective strategy to dodge the complications of motogen/mitogen receptor regulation however this overlooks the possibility of a corrective role behind signal desensitization in the correct guidance of cells in situ. As demonstrated by Minina et al. (2007), when CXCR4 receptor for a potent chemokine, stromal derived factor-1 (SDF-1) is constitutively expressed on zebrafish primordial germ cells, PGCs show ectopic chemotaxis towards multiple physiologically 42 incorrect sources of SDF-1. Therefore it is possible that our results reflect extended receptor desensitization or ligand degradation resulting in the cell maintaining a homeostatic level of intracellular signaling contributing to migration and therefore little to no change in cell motility. In contrast, direct current EF simulation dramatically increases cell motility and migration directionality compared to a chemical stimulant alone. It has the surprising effect of not only directing cell migration en mass towards the same general direction, but also increasing migration speed and net distance travelled. Previous studies of cell migration in electric fields have demonstrated that the phenomenon does not stem from an artificial electrophoretic ligand concentration gradient (Babona-Pilipos et. al, 2011) though it doesn’t necessarily rule out that this may contribute in part to the increase in directedness or motility. By exposing cells to an electric field, many intracellular signaling intermediates and surface receptors are concentrated in one direction. This includes Rac, Rho kinase and other cytoskeleton regulators that determine form the leading edge. It also establishes a persistent cell polarity in which secretory vesicles and actin polymers can be concentration. Conversely, exposure to a uniform chemical ligand concentration presents a uniform global activation of surface receptors. Instead of a chemotactic gradient which provides a directional cue to establish a differential level of signaling from the leading vs. the lagging edge, signal cascades are fired from all directions, preventing the formation of a dominant polarity in one direction. It is analogous to a cell which travels in one committed direction compared to a cell receiving signals from all ends and constantly changing directions. A similar behavior was reported by Pankov et.al. (2005) that reported suppression of Rac activity lead to formation of single dominant protrusions that lead cell migration. Conversely, global Rac activation, likely by uniform growth factor or purinergic receptor induces formation of many peripheral protrusions competing for cell polarity (Pankov, 2005). 43 However when combining these two modalities, we observed the directionality of NSC electrotaxis is significantly higher. Previous studies demonstrated electric fields can polarize surface membrane receptor (Zhao, 2009). One current hypothetical model demonstrates the alignment of chemical receptors along the direction of the electric field which may explain the observance of increased directedness of migration, and it is therefore possible that activation of a concentration of receptors on one side of the cell may increase directedness when combined (Wu and Lin, 2011). There were not sufficient repetitions to validate if this trend is a validated phenomenon the direction of results appears consistent. Unfortunately there is no data for bFGF and ATP-EF stimulation conditions due to restrictions of time and resources. Though because of the similar role these cytokines play, a similar trend in increasing migration directionality suggests a common intracellular role for motility factors in electric field. Possibly in upregulating mechanisms that promote cell polarity, amplifying intracellular signaling in response to cell’s detection of an electric field or actin cytoskeleton dynamics that are distinct from those involved in motility. One of the most important lessons learned from this of this project was how dynamic pure cell population can be even under consistent culture conditions and media. The NSCs provided in this study were generously donated from Dr. Jing Liu, who oversees the differentiation of H9 embryonic stem cells to NSCs. Many pilot and data gathering experiments, both successful and otherwise, were completed with these cells but they were by no means a consistent cell line. Stable cell or tumor cell lines proliferate quickly, produce cells that respond consistently between passages events and quite resistant to changes environment. NSCs on the other hand are by experience particularly susceptible to in vitro culture conditions. To maintain 44 NSC cell state in vitro, cultures must be passaged periodically to prevent overgrowth and cell senescence. The process of passage is usually quite traumatic to NSCs, killing at least 10% with each pass. For each progressive pass, the individual surviving cells can over time accumulate cell damage that changes proliferation and migration behavior. Based on experience, cells at later passages tend to have slightly different morphologies and are more difficult to passage additional times. To avoid the possibility of differentiation or genetic mutations due to repeated cell damage, the number of passages acceptable for experiments was kept within a narrow range. NSCs were received, at the earliest, four passage events after differentiation and all experiments were performed with cells between 6-10 passages. Cell motility and proliferation patterns appear to change between passage numbers and this is evident in the experimental results by the varying baseline speeds between experiments. Likewise the timing of the culture period from the day of passage to the day of experimentation was also relatively rigid. To promote survival and proliferation following a passage, cells are usually plated at a high density to allow paracrine and autocrine survival signaling. This is usually effective at recovering cells during culturing phase. The consequence of plating cells at high density is the time between passages to 100% culture confluence is relatively short. Whereas a stable cell line can be plated at very low densities, less than 10% confluence, and still be expected to grow for several days before reaching confluence. It required a balance act of maintaining cell cultures long enough to grow enough cells for future experiments but not long enough to collect cells when they are beyond the log phase of their growth curve. The timing of this phase also varied as passage numbers accumulated. Minor changes in the passage event were also found to change migration behavior during time lapse recording. During culture period, cells can recover post passage over several days and by and 45 negative effects from the passage are not seen. As little as 30 seconds longer during digestion period or cell dissociation by pipetting could dramatically change cell motility. With less time to recover from disturbances, the difference between cultures becomes more apparent. Another important technical aspect learned during this project is the importance of physical environmental factors on cells when the scale becomes increasingly small. During an experiment, cell density is intentionally low for purposes of individual cell quantification to minimize intercellular interactions that introduce another variable factor. Unfortunately, higher culture densities that provide greater cell-cell contact promote survivability and tolerance to environmental changes such as media temperature and flow. Larger media volumes used in culture are also better at buffering physical changes. Whereas in an experiment changing media during an experiment it also has the unwanted side effect of creating fluid flow of only a few milliliters of liquid through an opening .1mm2 big. These were similar concerns regarding pH change, physical vibrations and even light exposure. Though the scale of our model does not deal in microliter volumes, the technical difficulties of this model mimics those found when working with microfluidic devices. Technical complications aside, the relevance of the aforementioned project rests on the implications of the findings to an in vivo setting. Though the in vitro experimental system is biologically minimal, it is also somewhat of a privileged condition for cell migration. It lacks the same cytotoxic conditions, inflammatory response and recruitment of innate immune system components to the injured brain and is therefore is less stressful compared to an in vivo postinjury environment. Additionally, migration of cells on an unobstructed 2D surface is significantly different compared to 3D tissue network of extracellular matrix and interconnected 46 cells. However in vitro migration assays can demonstrate the basic cellular response using a simplified stimulation construct. It is known that electrical and chemical gradients and cues have general concurrent roles in directing mass cell migration behind tissue regeneration and also early embryonic development. In this manner, we can investigate the response level that can be attributed to each type of stimuli. In our studies here, the preliminary data presented suggests that a chemical and electrical co-stimulation method may enhance the efficiency of EF guided NSC migration. Though we did not see an additive or sustained increase in motility, it suggests that once optimized, EGF plays a role in increasing directedness. Conversely, it suggests that with an optimized administration of EGF, a lower EF strength can be applied in order to achieve the same level of directed migration. This model also only represents two classes of stimuli in a process that involves many other factors affecting tissue repair/formation forces. 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