| Literature DB >> 32043565 |
Ana Sánchez-Monteagudo1,2, María Álvarez-Sauco3, Isabel Sastre4, Irene Martínez-Torres4, Vincenzo Lupo1,2, Marina Berenguer2,5,6, Carmen Espinós1,2.
Abstract
Wilson's disease (WD) is an autosomal recessive disorder caused by ATP7B mutations. Subjects with only one mutation may show clinical signs and individuals with biallelic changes may remain asymptomatic. We aimed to achieve a conclusive genetic diagnosis for 34 patients clinically diagnosed of WD. Genetic analysis comprised from analysis of exons to WES (whole exome sequencing), including promoter, introns, UTRs (untranslated regions), besides of study of large deletions/duplications by MLPA (multiplex ligation-dependent probe amplification). Biallelic ATP7B mutations were identified in 30 patients, so that four patients were analyzed using WES. Two affected siblings resulted to be compound heterozygous for mutations in CCDC115, which is involved in a form of congenital disorder of glycosylation. In sum, the majority of patients with a WD phenotype carry ATP7B mutations. However, if genetic diagnosis is not achieved, additional genes should be considered because other disorders may mimic WD.Entities:
Keywords: zzm321990ATP7B gene; zzm321990CCDC115 gene; Wilson's disease; Wilson-like phenotype; genetic diagnosis; targeted next-generation sequencing; whole exome sequencing
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Year: 2020 PMID: 32043565 DOI: 10.1111/cge.13719
Source DB: PubMed Journal: Clin Genet ISSN: 0009-9163 Impact factor: 4.438