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DOI 10.3390/cells14241985

Медицинская библиотека · Cells · 2025

Karyotypic Profiling of Induced Pluripotent Stem Cells Derived from a Xeroderma Pigmentosum Group C Patient.

Alsalloum Almaqdad, Mingaleva Natalia, Gornostal Ekaterina, Antysheva Zoia, Sparber Peter, Skoblov Mikhail, Pozhitnova Victoria, Belysheva Tatiana, Levashova Aygun, Kuznetsova Ekaterina, Suvorova Yulia, Krupinova Julia, Bogdanov Viktor, Abyzov Alexej, Mityaeva Olga, Volchkov Pavel

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Аннотация

Xeroderma Pigmentosum group C (XP-C) is an autosomal recessive disorder caused by mutations in the XPC gene, leading to defective nucleotide excision repair. This defect leads to genomic instability and a profound cancer predisposition. To model this disease, we generated induced pluripotent stem cells (iPSCs) from an XP-C patient carrying a novel homozygous nonsense mutation in the XPC gene (c.1830C>A). The resulting iPSCs demonstrated typical pluripotent characteristics, including expression of key markers and trilineage differentiation capability. However, genomic assessment revealed progressive karyotypic instability during extended culture. While initial whole-genome sequencing detected no major chromosomal abnormalities, subsequent G-banding analysis identified acquired trisomy 12 in two lines (CL12 and CL27) and a derivative X chromosome in a third line (CL30). These abnormalities were absent in early-passage analyses, indicating that they were acquired and selected for during extended culture. The acquisition of a derivative X chromosome in CL30, alongside recurrent trisomy 12, represents a unique cytogenetic signature likely attributable to the underlying XPC defect. We hypothesize that the loss of GG-NER creates a permissive genomic environment, accelerating the accumulation of DNA damage and chromosomal missegregation under replicative stress. This temporal divergence in genomic integrity highlights how culture pressures drive chromosomal evolution in XP-C iPSCs independently of initial reprogramming. Our findings emphasize that XP-C iPSCs require continuous genomic surveillance and provide a model for investigating how DNA repair deficiencies interact with in vitro culture stress.

Для цитирования: Alsalloum Almaqdad, Mingaleva Natalia, Gornostal Ekaterina, Antysheva Zoia, Sparber Peter, Skoblov Mikhail, Pozhitnova Victoria, Belysheva Tatiana, Levashova Aygun, Kuznetsova Ekaterina, Suvorova Yulia, Krupinova Julia, Bogdanov Viktor, Abyzov Alexej, Mityaeva Olga, Volchkov Pavel. Karyotypic Profiling of Induced Pluripotent Stem Cells Derived from a Xeroderma Pigmentosum Group C Patient.. Cells. 2025. DOI: 10.3390/cells14241985.

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