Production of an induced pluripotent stem cell line CSSi018-A (14192) from a patient with hypomyelinating leukodystrophy 7 (HLD7) carrying biallelic variants of POLR3A (c.1802 T > A; c.4072G > A)

Stem Cell Res. 2024 Aug:78:103468. doi: 10.1016/j.scr.2024.103468. Epub 2024 Jun 6.

Abstract

Hypomyelinating leukodystrophies (HLD) are a group of heterogeneous genetic disorders characterized by a deficit in myelin deposition during brain development. Specifically, 4H-Leukodystrophy is a recessive disease due to biallelic mutations in the POLR3A gene, which encodes one of the subunits forming the catalytic core of RNA polymerase III (PolIII). The disease also presents non-neurological signs such as hypodontia and hypogonadotropic hypogonadism. Here, we report the generation of a human induced pluripotent stem cell (hiPSC) line from fibroblasts of the first identified carrier of the biallelic POLR3A variants c.1802 T > A and c.4072G > A.

MeSH terms

  • Alleles
  • Cell Line
  • Hereditary Central Nervous System Demyelinating Diseases / genetics
  • Hereditary Central Nervous System Demyelinating Diseases / pathology
  • Humans
  • Induced Pluripotent Stem Cells* / metabolism
  • Male
  • RNA Polymerase III* / genetics
  • RNA Polymerase III* / metabolism

Substances

  • RNA Polymerase III
  • POLR3A protein, human