MedElevator Войти в исследования
← Поиск публикаций

DOI 10.3390/ijms232314965

Медицинская библиотека · International journal of molecular sciences · 2022

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

СтатьяПеревод в очереди

Аннотация

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.

Для цитирования: 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. Congenital Stationary Night Blindness: Clinical and Genetic Features.. International journal of molecular sciences. 2022. DOI: 10.3390/ijms232314965.

Предметные рубрики MeSH

Подготовка и перевод публикации
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…

История · было → стало

Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.

Тип публикации указан по данным источника. Уровень доказательности автоматически не присваивается.