Generation of the induced pluripotent stem cell line ISMMSi061-A from a patient with ataxia, intention tremor, and hypotonia syndrome, childhood-onset
We have described a novel neurodevelopmental disorder caused by de novo, heterozygous pathogenic variants in the Pou domain, class 4, transcription factor 1 (POU4F1), now termed ataxia, intention tremor, and hypotonia syndrome, childhood-onset (ATITHS). This study reports the generation and characterization of an induced pluripotent stem cell (iPSC) line derived from peripheral blood mononuclear cells of a patient with ATITHS. The resulting iPSC line, ISMMSi061-A, harbors the POU4F1 heterozygous variant c.271_281del;p.(Thr91Hisfs*254). The iPSCs display normal cell morphology, expression of pluripotency markers, normal karyotype, and the ability to differentiate into all three germ layers.
Authors
- Bryn D. Webb (ORCID: https://orcid.org/0000-0001-6174-4677)
- S. Salemi
- Erdene Baljinnyam
- Samuel Hubbard
- Norman N. Liu
- Samuele G. Marro
- Genevieve Smith
- Priya Pradhan
- Ruiqi Hu
Institutions
- Allen Institute for Brain Science (US)
- Pediatrics and Genetics (US)
- Icahn School of Medicine at Mount Sinai (US)
Publication Details
- Journal
- Stem Cell Research
- Published
- 2026-02-17
- DOI
- https://doi.org/10.1016/j.scr.2026.103938
- Primary Topic
- Genomics and Rare Diseases
- Type
- article
- Field-Weighted Citation Impact
- 0.00