从一例色盲患者的全基因组测序中鉴定出CNGB3致病性深内含子变异。

Case Reports in Genetics Pub Date : 2025-05-27 eCollection Date: 2025-01-01 DOI:10.1155/crig/3466358
Matthew R Gregory, Khurram Liaqat, Kayla Treat, Kathryn M Haider, Francesco Vetrini, Erin Conboy
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引用次数: 0

摘要

色盲(Achromatopsia,简称ACHM) (MIM: 262300)是一种常染色体隐性遗传病,以视力下降和色盲为特征。在此报告中,我们回顾了一例14岁男性色盲患者,他有视网膜营养不良史,视锥功能障碍,ERG正常的黑暗适应反应,先天性眼球震颤,远视和散光。诊断性外显子组测序先前显示单个母系遗传致病性CNGB3变异(c.1148delC, p.(T383lfs * 13))。在印第安纳大学医学院(usm)未确诊罕见病诊所(URDC)登记入组后,基因组测序(GS)发现了第二个CNGB3已知变异c.1663-1205G > a p.(Gly555Leufs∗33),该变异被归类为可能致病。在患者身上发现这种变异提供了分子诊断所需的证据,并结束了患者及其家属长达15年的诊断历程。一旦确诊,患者就有资格接受基因治疗,并有资格接受国家职业康复计划和生物驾驶。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Pathogenic Deep Intronic Variant in CNGB3 Identified From Whole-Genome Sequencing in an Unsolved Case of Patient Affected With Achromatopsia.

Achromatopsia (ACHM) (MIM: 262300) is an autosomal recessive disorder characterized by reduced visual acuity and color blindness. In this report, we review the case of a 14-year-old male patient diagnosed with achromatopsia with a history of retinal dystrophy, cone dysfunction with normal dark-adapted response on ERG, congenital nystagmus, farsightedness, and astigmatism. The diagnostic exome sequencing previously revealed a single maternally inherited pathogenic CNGB3 variant (c.1148delC, p.(T383lfs∗13). Following enrollment in the Undiagnosed Rare Disease Clinic (URDC) at Indiana University School of Medicine (IUSM), genome sequencing (GS) identified a second CNGB3 known variant c.1663-1205G > A p.(Gly555Leufs∗33), which was classified as likely pathogenic. Identification of this variant in the patient provided the evidence needed for a molecular diagnosis and ended a 15-year diagnostic odyssey for the patient and his family. With a diagnosis, the patient is eligible for gene therapy and qualifies for the state-run Vocational Rehabilitation Program and bioptic driving.

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