| Literature DB >> 33669941 |
Nina Kobal1, Tjaša Krašovec1, Maja Šuštar1, Marija Volk2, Borut Peterlin2, Marko Hawlina1, Ana Fakin1.
Abstract
Mutations in rhodopsin gene (RHO) are a frequent cause of retinitis pigmentosa (RP) and less often, congenital stationary night blindness (CSNB). Mutation p.G90D has previously been associated with CSNB based on the examination of one family. This study screened 60 patients. Out of these 60 patients, 32 were affected and a full characterization was conducted in 15 patients. We described the clinical characteristics of these 15 patients (12 male, median age 42 years, range 8-71) from three families including visual field (Campus Goldmann), fundus autofluorescence (FAF), optical coherence tomography (OCT) and electrophysiology. Phenotypes were classified into four categories: CSNB (N = 3, 20%) sector RP (N = 3, 20%), pericentral RP (N = 1, 6.7%) and classic RP (N = 8, 53.3% (8/15)). The phenotypes were not associated with family, sex or age (Kruskal-Wallis, p > 0.05), however, cystoid macular edema (CME) was observed only in one family. Among the subjects reporting nyctalopia, 69% (22/32) were male. The clinical characteristics of the largest p.G90D cohort so far showed a large frequency of progressive retinal degeneration with 53.3% developing RP, contrary to the previous report.Entities:
Keywords: CSNB; ERG; FAF; G90D; OCT; RHO; RP; congenital stationary night blindness; constitutively active mutation; electroretinography; fundus autofluorescence; inherited retinal dystrophy; pericentral retinitis pigmentosa; retinal degeneration; retinitis pigmentosa; rhodopsin; sector retinitis pigmentosa
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Year: 2021 PMID: 33669941 PMCID: PMC7924842 DOI: 10.3390/ijms22042133
Source DB: PubMed Journal: Int J Mol Sci ISSN: 1422-0067 Impact factor: 5.923