Litcius/Paper detail

High-level correction of the sickle mutation is amplified in vivo during erythroid differentiation

Wendy Magis, Mark A. DeWitt, Stacia K. Wyman, Jonathan T. Vu, Seok‐Jin Heo, Shirley Shao, Finn Hennig, Zulema Romero, Beatriz Campo-Fernández, Suzanne Saïd, Matthew McNeill, Garrett R. Rettig, Yongming Sun, Yu Wang, Mark A. Behlke, Donald B. Kohn, Dario Boffelli, Mark C. Walters, Jacob E. Corn, David I. K. Martin

2022iScience51 citationsDOIOpen Access PDF

Abstract

Background: A point mutation in sickle cell disease (SCD) alters one amino acid in the β-globin subunit of hemoglobin, with resultant anemia and multiorgan damage that typically shortens lifespan by decades. Because SCD is caused by a single mutation, and hematopoietic stem cells (HSCs) can be harvested, manipulated, and returned to an individual, it is an attractive target for gene correction. Results: erythroid differentiation markedly enriches for corrected β-globin alleles, indicating that erythroblasts carrying one or more corrected alleles have a survival advantage. Significance: These findings indicate that the sickle mutation can be corrected in autologous HSCs with an optimized protocol suitable for clinical translation.

Topics & Concepts

GlobinBiologyHaematopoiesisMutationPoint mutationFetal hemoglobinStem cellIn vivoGeneticsHemoglobinAlleleSickle cell anemiaMolecular biologyCell biologyGeneCellBiochemistryPregnancyFetusHemoglobinopathies and Related DisordersAcute Myeloid Leukemia ResearchGenomics and Rare Diseases