Literature DB >> 33669941

Stationary and Progressive Phenotypes Caused by the p.G90D Mutation in Rhodopsin Gene.

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

Mesh:

Substances:

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


  51 in total

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Review 2.  Congenital stationary night blindness: an analysis and update of genotype-phenotype correlations and pathogenic mechanisms.

Authors:  Christina Zeitz; Anthony G Robson; Isabelle Audo
Journal:  Prog Retin Eye Res       Date:  2014-10-13       Impact factor: 21.198

Review 3.  ISCEV standard for clinical multifocal electroretinography (mfERG) (2011 edition).

Authors:  Donald C Hood; Michael Bach; Mitchell Brigell; David Keating; Mineo Kondo; Jonathan S Lyons; Michael F Marmor; Daphne L McCulloch; Anja M Palmowski-Wolfe
Journal:  Doc Ophthalmol       Date:  2011-10-30       Impact factor: 2.379

4.  Different amino acid substitutions at the same position in rhodopsin lead to distinct phenotypes.

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Journal:  Invest Ophthalmol Vis Sci       Date:  2006-04       Impact factor: 4.799

5.  Fundus autofluorescence and ellipsoid zone (EZ) line width can be an outcome measurement in RHO-associated autosomal dominant retinitis pigmentosa.

Authors:  Vitor K L Takahashi; Júlia T Takiuti; Jose R L Carvalho-Jr; Christine L Xu; Jimmy K Duong; Vinit B Mahajan; Stephen H Tsang
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2019-01-11       Impact factor: 3.117

6.  CLINICAL CHARACTERISTICS AND NATURAL HISTORY OF RHO-ASSOCIATED RETINITIS PIGMENTOSA: A Long-Term Follow-Up Study.

Authors:  Xuan-Thanh-An Nguyen; Mays Talib; Caroline van Cauwenbergh; Mary J van Schooneveld; Marta Fiocco; Jan Wijnholds; Jacoline B Ten Brink; Ralph J Florijn; Nicoline E Schalij-Delfos; Gislin Dagnelie; Maria M van Genderen; Elfride de Baere; Magda A Meester-Smoor; Julie De Zaeytijd; Irina Balikova; Alberta A Thiadens; Carel B Hoyng; Caroline C Klaver; L Ingeborgh van den Born; Arthur A Bergen; Bart P Leroy; Camiel J F Boon
Journal:  Retina       Date:  2021-01-01       Impact factor: 4.256

7.  Retinal degeneration in mice expressing the constitutively active G90D rhodopsin mutant.

Authors:  Alejandro T Colozo; Sreelakshmi Vasudevan; Paul S-H Park
Journal:  Hum Mol Genet       Date:  2020-04-15       Impact factor: 6.150

8.  Clinical and Genetic Analysis of a European Cohort with Pericentral Retinitis Pigmentosa.

Authors:  Marianthi Karali; Francesco Testa; Raffaella Brunetti-Pierri; Valentina Di Iorio; Mariateresa Pizzo; Paolo Melillo; Maria Rosaria Barillari; Annalaura Torella; Francesco Musacchia; Luigi D'Angelo; Sandro Banfi; Francesca Simonelli
Journal:  Int J Mol Sci       Date:  2019-12-20       Impact factor: 5.923

9.  Genetic landscape of 6089 inherited retinal dystrophies affected cases in Spain and their therapeutic and extended epidemiological implications.

Authors:  Irene Perea-Romero; Gema Gordo; Ionut F Iancu; Marta Corton; Carmen Ayuso; Marta Del Pozo-Valero; Berta Almoguera; Fiona Blanco-Kelly; Ester Carreño; Belen Jimenez-Rolando; Rosario Lopez-Rodriguez; Isabel Lorda-Sanchez; Inmaculada Martin-Merida; Lucia Pérez de Ayala; Rosa Riveiro-Alvarez; Elvira Rodriguez-Pinilla; Saoud Tahsin-Swafiri; Maria J Trujillo-Tiebas; Blanca Garcia-Sandoval; Pablo Minguez; Almudena Avila-Fernandez
Journal:  Sci Rep       Date:  2021-01-15       Impact factor: 4.379

10.  Variant detection sensitivity and biases in whole genome and exome sequencing.

Authors:  Alison M Meynert; Morad Ansari; David R FitzPatrick; Martin S Taylor
Journal:  BMC Bioinformatics       Date:  2014-07-19       Impact factor: 3.169

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  1 in total

1.  Disease Progression in CNGA3 and CNGB3 Retinopathy; Characteristics of Slovenian Cohort and Proposed OCT Staging Based on Pooled Data from 126 Patients from 7 Studies.

Authors:  Manca Tekavčič Pompe; Nika Vrabič; Marija Volk; Andrej Meglič; Martina Jarc-Vidmar; Borut Peterlin; Marko Hawlina; Ana Fakin
Journal:  Curr Issues Mol Biol       Date:  2021-08-16       Impact factor: 2.976

  1 in total

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