Neveu Magella M, Padhy Srikanta Kumar, Ramamurthy Srishti, Takkar Brijesh, Jalali Subhadra, Cp Deepika, Padhi Tapas Ranjan, Robson Anthony G
Department Electrophysiology, Moorfields Eye Hospital, London, EC1V 2PD, UK.
Institute of Ophthalmology, University College London, London, UK.
Clin Ophthalmol. 2022 May 24;16:1569-1587. doi: 10.2147/OPTH.S329282. eCollection 2022.
Albinism describes a heterogeneous group of genetically determined disorders characterized by disrupted synthesis of melanin and a range of developmental ocular abnormalities. The main ocular features common to both oculocutaneous albinism (OCA), and ocular albinism (OA) include reduced visual acuity, refractive errors, foveal hypoplasia, congenital nystagmus, iris and fundus hypopigmentation and visual pathway misrouting, but clinical signs vary and there is phenotypic overlap with other pathologies. This study reviews the prevalence, genetics and ocular manifestations of OCA and OA, including abnormal development of the optic chiasm. The role of visual electrophysiology in the detection of chiasmal dysfunction and visual pathway misrouting is emphasized, highlighting how age-associated changes in visual evoked potential (VEP) test results must be considered to enable accurate diagnosis, and illustrated further by the inclusion of novel VEP data in genetically confirmed cases. Differential diagnosis is considered in the context of suspected retinal and other disorders, including rare syndromes that may masquerade as albinism.
白化病描述了一组由基因决定的异质性疾病,其特征是黑色素合成中断以及一系列眼部发育异常。眼皮肤白化病(OCA)和眼部白化病(OA)共有的主要眼部特征包括视力下降、屈光不正、黄斑发育不全、先天性眼球震颤、虹膜和眼底色素减退以及视觉通路错向,但临床症状存在差异,且与其他病症存在表型重叠。本研究回顾了OCA和OA的患病率、遗传学及眼部表现,包括视交叉的异常发育。强调了视觉电生理在检测视交叉功能障碍和视觉通路错向中的作用,突出了必须考虑视觉诱发电位(VEP)测试结果中与年龄相关的变化以实现准确诊断,并通过纳入基因确诊病例的新VEP数据进一步说明。在疑似视网膜疾病和其他病症的背景下考虑鉴别诊断,包括可能伪装成白化病的罕见综合征。