A patient-specific induced pluripotent stem cell and neural stem cell resource for angelman syndrome with 15q11.2-q13 deletion

Angelman syndrome (AS) is a severe neurodevelopmental disorder most commonly caused by a maternal deletion of the 15q11.2-q13 region, resulting in loss of ubiquitin protein ligase E3A ( UBE3A ) function. The development of targeted therapies and a deeper understanding of AS pathogenesis require the availability of genetically accurate human models. Here, we generated and characterized a novel induced pluripotent stem cell (iPSC) line from peripheral blood mononuclear cells of a female pediatric patient with genetically confirmed deletion-type AS. The iPSC line displayed a normal karyotype, expressed key pluripotency markers, and demonstrated trilineage differentiation potential. Efficient generation of neural stem cells establishes a foundational cellular resource for future investigations of neuronal maturation and disease-specific phenotypes. This well-validated iPSC and NSC resource provides a valuable and clinically relevant platform for elucidating the molecular pathophysiology of AS in a genetically accurate human cellular context and for high-throughput screening of therapeutic compounds targeting the predominant population of patients with AS.

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Publication Details

Journal
Scientific Reports
Published
2026-06-13
DOI
https://doi.org/10.1038/s41598-026-57826-2
Primary Topic
Genetic Syndromes and Imprinting
Type
article
Field-Weighted Citation Impact
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article

A patient-specific induced pluripotent stem cell and neural stem cell resource for angelman syndrome with 15q11.2-q13 deletion

Song Qu, Song Cui, Yanyan Wang, Pu Sun et al.
Scientific Reports
Genetic Syndromes and Imprinting
article

A patient-specific induced pluripotent stem cell and neural stem cell resource for angelman syndrome with 15q11.2-q13 deletion

Song Qu, Song Cui, Yanyan Wang, Pu Sun, Y J Wu
article en

Abstract

Angelman syndrome (AS) is a severe neurodevelopmental disorder most commonly caused by a maternal deletion of the 15q11.2-q13 region, resulting in loss of ubiquitin protein ligase E3A ( UBE3A ) function. The development of targeted therapies and a deeper understanding of AS pathogenesis require the availability of genetically accurate human models. Here, we generated and characterized a novel induced pluripotent stem cell (iPSC) line from peripheral blood mononuclear cells of a female pediatric patient with genetically confirmed deletion-type AS. The iPSC line displayed a normal karyotype, expressed key pluripotency markers, and demonstrated trilineage differentiation potential. Efficient generation of neural stem cells establishes a foundational cellular resource for future investigations of neuronal maturation and disease-specific phenotypes. This well-validated iPSC and NSC resource provides a valuable and clinically relevant platform for elucidating the molecular pathophysiology of AS in a genetically accurate human cellular context and for high-throughput screening of therapeutic compounds targeting the predominant population of patients with AS.

Scientific Reports
Army Medical University (CN), Children's Hospital of Chongqing Medical University (CN), Chongqing Medical University (CN)
Openalex Percentile: Top 6%
Genetic Syndromes and Imprinting
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