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Congenital Stationary Night Blindness: Clinical and Genetic Features.
Kim, Angela H; Liu, Pei-Kang; Chang, Yin-Hsi; Kang, Eugene Yu-Chuan; Wang, Hung-Hsuan; Chen, Nelson; Tseng, Yun-Ju; Seo, Go Hun; Lee, Hane; Liu, Laura; Chao, An-Ning; Chen, Kuan-Jen; Hwang, Yih-Shiou; Wu, Wei-Chi; Lai, Chi-Chun; Tsang, Stephen H; Hsiao, Meng-Chang; Wang, Nan-Kai.
Afiliación
  • Kim AH; Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Medical Center, New York, NY 10032, USA.
  • Liu PK; Downstate Medical Center, College of Medicine, State University of New York, Brooklyn, NY 11203, USA.
  • Chang YH; Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Medical Center, New York, NY 10032, USA.
  • Kang EY; Department of Ophthalmology, Kaohsiung Medical University Hospital, Kaohsiung Medical University, Kaohsiung 80756, Taiwan.
  • Wang HH; School of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung 80708, Taiwan.
  • Chen N; Institute of Biomedical Sciences, National Sun Yat-sen University, Kaohsiung 80424, Taiwan.
  • Tseng YJ; Department of Ophthalmology, Chang Gung Memorial Hospital, Linkou Medical Center, Taoyuan 33305, Taiwan.
  • Seo GH; College of Medicine, Chang Gung University, Taoyuan 33303, Taiwan.
  • Lee H; Department of Ophthalmology, Chang Gung Memorial Hospital, Linkou Medical Center, Taoyuan 33305, Taiwan.
  • Liu L; College of Medicine, Chang Gung University, Taoyuan 33303, Taiwan.
  • Chao AN; Graduate Institute of Clinical Medical Sciences, College of Medicine, Chang Gung University, Taoyuan 33303, Taiwan.
  • Chen KJ; Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Medical Center, New York, NY 10032, USA.
  • Hwang YS; Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Medical Center, New York, NY 10032, USA.
  • Wu WC; Faculty of Health Sciences, Queen's University, Kingston, ON K7L 3N6, Canada.
  • Lai CC; Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Medical Center, New York, NY 10032, USA.
  • Tsang SH; Division of Medical Genetics, 3billion, Inc., Seoul 06193, Korea.
  • Hsiao MC; Division of Medical Genetics, 3billion, Inc., Seoul 06193, Korea.
  • Wang NK; Department of Ophthalmology, Chang Gung Memorial Hospital, Linkou Medical Center, Taoyuan 33305, Taiwan.
Int J Mol Sci ; 23(23)2022 Nov 29.
Article en En | MEDLINE | ID: mdl-36499293
Congenital stationary night blindness (CSNB) is an inherited retinal disease (IRD) that causes night blindness in childhood with heterogeneous genetic, electrophysical, and clinical characteristics. The development of sequencing technologies and gene therapy have increased the ease and urgency of diagnosing IRDs. This study describes seven Taiwanese patients from six unrelated families examined at a tertiary referral center, diagnosed with CSNB, and confirmed by genetic testing. Complete ophthalmic exams included best corrected visual acuity, retinal imaging, and an electroretinogram. The effects of identified novel variants were predicted using clinical details, protein prediction tools, and conservation scores. One patient had an autosomal dominant CSNB with a RHO variant; five patients had complete CSNB with variants in GRM6, TRPM1, and NYX; and one patient had incomplete CSNB with variants in CACNA1F. The patients had Riggs and Schubert-Bornschein types of CSNB with autosomal dominant, autosomal recessive, and X-linked inheritance patterns. This is the first report of CSNB patients in Taiwan with confirmed genetic testing, providing novel perspectives on molecular etiology and genotype-phenotype correlation of CSNB. Particularly, variants in TRPM1, NYX, and CACNA1F in our patient cohort have not previously been described, although their clinical significance needs further study. Additional study is needed for the genotype-phenotype correlation of different mutations causing CSNB. In addition to genetic etiology, the future of gene therapy for CSNB patients is reviewed and discussed.
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Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedades Hereditarias del Ojo / Ceguera Nocturna / Enfermedades Genéticas Ligadas al Cromosoma X / Miopía Tipo de estudio: Diagnostic_studies / Prognostic_studies Límite: Humans Idioma: En Revista: Int J Mol Sci Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedades Hereditarias del Ojo / Ceguera Nocturna / Enfermedades Genéticas Ligadas al Cromosoma X / Miopía Tipo de estudio: Diagnostic_studies / Prognostic_studies Límite: Humans Idioma: En Revista: Int J Mol Sci Año: 2022 Tipo del documento: Article País de afiliación: Estados Unidos