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.
Authors
- Maddalena Di Nardo (ORCID: https://orcid.org/0000-0002-1481-2101)
- Marianna Paulis (ORCID: https://orcid.org/0000-0001-7803-9982)
- Antonio Musio (ORCID: https://orcid.org/0000-0001-7701-6543)
- S. P. Lucia (ORCID: https://orcid.org/0000-0003-3368-2135)
- Angela La Grua
Institutions
- 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)
Publication Details
- 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
- Field-Weighted Citation Impact
- 0.00