Literature DB >> 27627638

FUNDUS ALBIPUNCTATUS ASSOCIATED WITH CONE DYSFUNCTION.

Laura Kuehlewein1, Fadi Nasser, Nicola Gloeckle, Susanne Kohl, Eberhart Zrenner.   

Abstract

PURPOSE: To describe a case of cone dysfunction associated with fundus albipunctatus.
METHODS: This report is an observational case report. The examination included multimodal imaging, electrophysiological recordings after standard and prolonged dark adaption, and disease targeted gene panel sequencing.
RESULTS: In this report, the authors present a 55-year-old Chinese male with findings on fundus examination, optical coherence tomography, and full-field electroretinography after standard and prolonged dark adaption consistent with fundus albipunctatus associated with cone dysfunction. Disease targeted gene panel sequencing revealed two heterozygous mutations in RDH5 (c.124C>T; p.Arg42Cys and c.500G>A; p.Arg167His).
CONCLUSION: The authors report the case of a patient with ophthalmic findings characteristic for cone dysfunction in the setting of genetically confirmed fundus albipunctatus.

Entities:  

Mesh:

Year:  2017        PMID: 27627638     DOI: 10.1097/ICB.0000000000000420

Source DB:  PubMed          Journal:  Retin Cases Brief Rep        ISSN: 1935-1089


  6 in total

1.  A large animal model of RDH5-associated retinopathy recapitulates important features of the human phenotype.

Authors:  Laurence M Occelli; Anahita Daruwalla; Samantha R De Silva; Paige A Winkler; Kelian Sun; Nathaniel Pasmanter; Andrea Minella; Janice Querubin; Leslie A Lyons; Anthony G Robson; Elise Heon; Michel Michaelides; Andrew R Webster; Krzysztof Palczewski; Ajoy Vincent; Omar A Mahroo; Philip D Kiser; Simon M Petersen-Jones
Journal:  Hum Mol Genet       Date:  2022-04-22       Impact factor: 5.121

2.  Measurement of dark adaptometry during ISCEV standard flash electroretinography.

Authors:  Jeff Rabin; Brooke Houser; Carolyn Talbert; Rue Patel
Journal:  Doc Ophthalmol       Date:  2017-10-03       Impact factor: 2.379

3.  Animals Models of Inherited Retinal Disease.

Authors:  Ala Moshiri
Journal:  Int Ophthalmol Clin       Date:  2021-07-01

4.  A spectral-domain optical coherence tomographic analysis of Rdh5-/- mice retina.

Authors:  Yuting Xie; Takayuki Gonome; Kodai Yamauchi; Natsuki Maeda-Monai; Reiko Tanabu; Sei-Ichi Ishiguro; Mitsuru Nakazawa
Journal:  PLoS One       Date:  2020-04-09       Impact factor: 3.240

5.  Novel variants in the RDH5 Gene in a Chinese Han family with fundus albipunctatus.

Authors:  Tianwei Qian; Qiaoyun Gong; Hangqi Shen; Caihua Li; Gao Wang; Xun Xu; Isabelle Schrauwen; Weijun Wang
Journal:  BMC Ophthalmol       Date:  2022-02-11       Impact factor: 2.209

Review 6.  Large Animal Models of Inherited Retinal Degenerations: A Review.

Authors:  Paige A Winkler; Laurence M Occelli; Simon M Petersen-Jones
Journal:  Cells       Date:  2020-04-03       Impact factor: 6.600

  6 in total

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