Generation of CRISPR/Cas9-edited human iPSC lines carrying homozygous and heterozygous SAMD9 p.I983S mutations

Stem Cell Res. 2024 Dec:81:103532. doi: 10.1016/j.scr.2024.103532. Epub 2024 Aug 12.

Abstract

Induced pluripotent stem cells (iPSCs) harboring patient derived SAMD9 mutation offer a unique platform to study the multi-organ involvement observed in this rare disease, referred to as myelodysplasia, infections, restriction of growth, adrenal hypoplasia, genital phenotypes, and enteropathy (MIRAGE) syndrome. The pluripotent nature of iPSCs allows in vitro differentiation into various somatic cell types representing multiple organ systems affected in SAMD9-mutated patients. Hence, in this paper, we present a CRISPR/Cas9-engineered iPSC model carrying SAMD9 c.2948T>G, p.I983S mutation previously reported in two patients with severe MIRAGE syndrome.

Keywords: Bone marrow failure; CRISPR/Cas9; Hematology; SAMD9; iPSC.

MeSH terms

  • CRISPR-Cas Systems*
  • Cell Line
  • Gene Editing
  • Heterozygote
  • Homozygote*
  • Humans
  • Induced Pluripotent Stem Cells* / metabolism
  • Intracellular Signaling Peptides and Proteins / genetics
  • Intracellular Signaling Peptides and Proteins / metabolism
  • Male
  • Mutation

Substances

  • SAMD9 protein, human
  • Intracellular Signaling Peptides and Proteins