De Novo or inherited

Hereditary angioedema (HAE) is a rare genetic disease, characterized by transient and self-limiting episodes of subcutaneous or submucosal swelling that spontaneously resolve within two to five days. The most common form of HAE, HAE-C1-INH, is caused by deleterious mutations in the SERPING1 gene, en...

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Detalles Bibliográficos
Autores: Batlle-Masó, Laura|||0000-0002-4209-176X, Perurena Prieto, Janire|||0000-0002-3048-1058, Viñas-Giménez, Laura|||0000-0003-2913-1154, Aguiló-Cucurull, Aina|||0000-0001-5622-778X, Fernández-Álvarez, Paula|||0000-0003-2695-3531, Gil-Serrano, Johana|||0000-0002-0673-7391, Guilarte, Mar|||0000-0001-7242-9584, Colobrán Oriol, Roger|||0000-0002-5964-536X
Tipo de recurso: artículo
Fecha de publicación:2025
País:España
Institución:Universitat Autònoma de Barcelona
Repositorio:Dipòsit Digital de Documents de la UAB
Idioma:inglés
OAI Identifier:oai:ddd.uab.cat:319417
Acceso en línea:https://ddd.uab.cat/record/319417
https://dx.doi.org/urn:doi:10.3389/fimmu.2025.1550380
Access Level:acceso abierto
Palabra clave:Hereditary angioedema
C1 inhibitor deficiency
SERPING1
Somatic variant
Gonosomal mosaicism
Genetic counseling
Descripción
Sumario:Hereditary angioedema (HAE) is a rare genetic disease, characterized by transient and self-limiting episodes of subcutaneous or submucosal swelling that spontaneously resolve within two to five days. The most common form of HAE, HAE-C1-INH, is caused by deleterious mutations in the SERPING1 gene, encoding the C1-Inhibitor protein, and its diagnosis is confirmed by decreased C1-INH function. Distinctively from other genetic forms of HAE, up to 15-20% of HAE-C1-INH cases are sporadic caused by de novo mutations. Here, we report a patient with apparently sporadic HAE-C1-INH. The patient had compatible clinical symptoms and a markedly low C1-INH function, and the parents showed normal values of C4 and normal C1-INH function. In the patient, we identified a novel splice site mutation in SERPING1 (c.890-1G.