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.

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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
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article

Generation of the induced pluripotent stem cell line ISMMSi061-A from a patient with ataxia, intention tremor, and hypotonia syndrome, childhood-onset

Bryn D. Webb, S. Salemi, Erdene Baljinnyam, Samuel Hubbard et al.
Stem Cell Research
Genomics and Rare Diseases
article

Generation of the induced pluripotent stem cell line ISMMSi061-A from a patient with ataxia, intention tremor, and hypotonia syndrome, childhood-onset

Bryn D. Webb, S. Salemi, Erdene Baljinnyam, Samuel Hubbard, Norman N. Liu, Samuele G. Marro, Genevieve Smith, Priya Pradhan, Ruiqi Hu
article en

Abstract

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.

Stem Cell ResearchVol. 92
Allen Institute for Brain Science (US), Pediatrics and Genetics (US), Icahn School of Medicine at Mount Sinai (US)
Openalex Percentile: Top 51%
Genomics and Rare Diseases
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