UNIVERSITY OF MALTA LIFE SCIENCE RESEARCH SEMINARS Haemoglobin

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UNIVERSITY OF MALTA

LIFE SCIENCE RESEARCH SEMINARS

Web: http://www.um.edu.mt/events/scisem/ Email: scisem@um.edu.mt

Abstract form

Title:

Models to Understand the Developmental Control of Foetal

Haemoglobin

Presenter:

Laura Grech

Contact address:

Tel:

79959025

Fax:

Email: lauragrech@yahoo.com

Presentation date:

7 th April 2014

Abstract

In this study we used three different available models to study the developmental control and regulation of HbF. These models included Haemoglobin F Malta I heterozygote (cords n=200; adults n=20), HPFH-KLF1 haploinsufficient family members (n=5) and Cell Models (primary human erythroid progenitor cells) (n=15). PCR genotyping for XMN1, BCL11A and HBS1L-MYB and

DNA sequencing for (AT)xTy and KLF1 was carried out on all samples. Erythroid progenitor cells were cultured from 15 donors (5 healthy adults, 5 thalassaemic homozygotes and 5 KLF1 haploinsufficient family members). Proliferating and differentiating cells were cultured under the effect of Thalidomide, 5’-Aza-2’-deoxycytidine and Hydroxyurea. The cell pellets extracted from these cultures on different culturing days were used for globin chain analysis and for RNA expression of globin and other trans -acting genes. When real-time qPCR was performed, an increa se in gamma globin and downregu l ation of KLF1 in the HEP s treated with 1μM thalidomide was noted in one thalassaemic homozygote patient carrying the IVSI-6C/COD39T mutation. An increase in gamma globin and decrease in KLF1 was also noted in three out of four t halassaemic patients. With hydro xyurea 10μM and 100μM the least responsive thalass a emic patient was the one carrying the long isoform of (AT)

X

T

Y

that is the (AT)

9

T

5

. All the other thalassaemic patients carried the short isoform of the (AT)

X

T

Y

that is the (AT)

7

T

7

. It is known that disruptive ATxTy (long form) give rise to an increase in β-globin bec ause BP1 is inhibited from binding. The data of all three models will be presented and discussed regarding to their connection on HbF regulation and control.

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