| Literature DB >> 29345414 |
Eulalie Lasseaux1, Claudio Plaisant1, Vincent Michaud1, Perrine Pennamen1,2, Aurelien Trimouille1,2, Laetitia Gaston1, Solène Monfermé3, Didier Lacombe1,2, Caroline Rooryck1,2, Fanny Morice-Picard4, Benoît Arveiler1,2.
Abstract
Albinism is a clinically and genetically heterogeneous disease characterized by variable degrees of hypopigmentation and by nystagmus, foveal hypoplasia, and chiasmatic misrouting of the optic nerves. The wide phenotypic heterogeneity impedes the establishment of phenotype-genotype correlations. To obtain a precise diagnosis, we screened the 19 known albinism genes in 990 index patients using targeted next-generation sequencing (NGS) and high-resolution comparative genomic hybridization. A molecular diagnosis was obtained in 72.32% of patients. A total of 243 new pathogenic variants were identified. Intragenic rearrangements represented 10.8% of all pathogenic alleles. NGS panel analysis allowed establishing a diagnosis for the rarest forms of the disease, which could not be diagnosed otherwise. Because of the clinical overlap between the different forms of the disease, diagnosis nowadays clearly relies on molecular grounds.Entities:
Keywords: albinism; deletions; mutations; next-generation sequencing
Mesh:
Year: 2018 PMID: 29345414 DOI: 10.1111/pcmr.12688
Source DB: PubMed Journal: Pigment Cell Melanoma Res ISSN: 1755-1471 Impact factor: 4.693