Generation and characterization of human iPSC lines from two patients with therapy-resistant epilepsy carrying nonsense heterozygous variants in the SMC1A gene

Different SMC1A variants contribute to a spectrum of phenotypes. Missense or small in-frame deletions are associated with Cornelia de Lange syndrome (CdLS) while SMC1A truncation variants have been detected in subjects with a clinical phenotype different from CdLS, with moderate-to-severe intellectual disability (ID) and pharmaco-resistant epilepsy. We generated two human iPSC lines from two patients with pharmaco-resistant epilepsy carrying nonsense heterozygous c.901C > T (p.E323*) and c.3103C > T (p.R1035*) variants in the SMC1A gene. These cell lines will be a valuable resource for in vitro disease modeling and drug testing for pharmaco-resistant epilepsy due to SMC1A variants.

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Journal
Stem Cell Research
Published
2025-06-16
DOI
https://doi.org/10.1016/j.scr.2025.103752
Primary Topic
Genetics and Neurodevelopmental Disorders
Type
article
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article

Generation and characterization of human iPSC lines from two patients with therapy-resistant epilepsy carrying nonsense heterozygous variants in the SMC1A gene

Maddalena Di Nardo, Marianna Paulis, Antonio Musio, S. P. Lucia et al.
Stem Cell Research
Genetics and Neurodevelopmental Disorders
article

Generation and characterization of human iPSC lines from two patients with therapy-resistant epilepsy carrying nonsense heterozygous variants in the SMC1A gene

Maddalena Di Nardo, Marianna Paulis, Antonio Musio, S. P. Lucia, Angela La Grua
article en

Abstract

Different SMC1A variants contribute to a spectrum of phenotypes. Missense or small in-frame deletions are associated with Cornelia de Lange syndrome (CdLS) while SMC1A truncation variants have been detected in subjects with a clinical phenotype different from CdLS, with moderate-to-severe intellectual disability (ID) and pharmaco-resistant epilepsy. We generated two human iPSC lines from two patients with pharmaco-resistant epilepsy carrying nonsense heterozygous c.901C > T (p.E323*) and c.3103C > T (p.R1035*) variants in the SMC1A gene. These cell lines will be a valuable resource for in vitro disease modeling and drug testing for pharmaco-resistant epilepsy due to SMC1A variants.

Stem Cell ResearchVol. 87
Istituto di Scienza e Tecnologie dell'Informazione "Alessandro Faedo" (IT), Institute of Genetic and Biomedical Research (IT), National Research Council (IT), IRCCS Humanitas Research Hospital (IT)
Openalex Percentile: Top 84%
Genetics and Neurodevelopmental Disorders
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