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DOI 10.3390/ijms26125762

Медицинская библиотека · International journal of molecular sciences · 2025

Clinical and Allelic Heterogeneity in a Small Cohort of Patients with Inherited Epidermolysis Bullosa.

Buianova Anastasiia A, Yagizarova Anastasia S, Kosykh Anastasiya V, Kubanov Alexey A, Belova Vera A, Shmitko Anna O, Karamova Arfenya E, Martynova Aleksandra A, Podmoskovnikov Grigoriy S, Nefedova Maria A, Monchakovskaya Ekaterina S, Korostin Dmitriy O, Gurskaya Nadya G, Rebrikov Denis V

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Аннотация

Inherited epidermolysis bullosa (EB) comprises a group of genetic disorders characterized by fragile skin that blisters easily. Targeted therapies for EB necessitate personalized approaches, underscoring the importance of precise diagnostics through genetic analysis and skin biopsy using transmission electron microscopy and/or immunohistochemistry. This study highlights the application of whole-exome sequencing (WES) to identify key pathogenic variants associated with EB. Most identified variants were associated with the recessive form of dystrophic EB, including four novel COL7A1 mutations: p.Leu1488ArgfsTer222, c.7759-3C>G, p.Gln1886Ter, and c.6501+6T>C, as well as recurrent variants p.Lys142Arg and p.Gly2049Glu. Additionally, variants were detected in KRT5 (c.971T>C, p.Val324Ala), associated with EB simplex, and in LAMB3 (c.2500C>T, p.Gln834Ter) in the homozygous state, associated with junctional EB. In silico splice prediction tools suggested disrupted splicing in both cases. One patient received topical gentamicin therapy targeting the nonsense mutation p.Gln1886Ter. These findings underscore the utility of WES in EB diagnostics, broaden the mutation spectrum, and contribute to the understanding of genotype-phenotype correlations in adult patients with EB.

Для цитирования: Buianova Anastasiia A, Yagizarova Anastasia S, Kosykh Anastasiya V, Kubanov Alexey A, Belova Vera A, Shmitko Anna O, Karamova Arfenya E, Martynova Aleksandra A, Podmoskovnikov Grigoriy S, Nefedova Maria A, Monchakovskaya Ekaterina S, Korostin Dmitriy O, Gurskaya Nadya G, Rebrikov Denis V. Clinical and Allelic Heterogeneity in a Small Cohort of Patients with Inherited Epidermolysis Bullosa.. International journal of molecular sciences. 2025. DOI: 10.3390/ijms26125762.

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