Hosseini Seyedeh Maryam, Nejad Shahrokh Abadi Reza, Babaei Meisam, Eghbal Fatemeh, Hashemi Narges
Eye Research Center Mashhad University of Medical Sciences Mashhad Iran.
Faculty of Medicine Mashhad University of Medical Sciences Mashhad Iran.
Clin Case Rep. 2024 Nov 15;12(11):e9566. doi: 10.1002/ccr3.9566. eCollection 2024 Nov.
This case report documents the experience of a 5-year-old girl who showed signs of retinal degeneration as the initial symptom of neuronal ceroid lipofuscinosis (NCLs). She originally presented with visual failure, which rapidly progressed to near total bilateral blindness. Two years later, she developed seizures and cognitive impairment, leading to a diagnosis of NCL7 resulting from a homozygote mutation in the MFSD8 gene. This case underscores the importance of considering NCLs as a potential diagnosis in cases of cone-rod dystrophy and visual loss as the primary clinical feature. It also emphasizes the early onset and initial presentation of retinal degeneration associated with NCL7, before other signs and symptoms manifest, as the second documented case of its kind. Due to the potential for NCL7 to present initially with visual loss before other hallmark signs, it is crucial to consider it among various syndromic and non-syndromic disorders in the differential diagnosis.
本病例报告记录了一名5岁女孩的经历,她最初表现出视网膜变性症状,这是神经元蜡样脂褐质沉积症(NCLs)的首发症状。她最初出现视力减退,并迅速发展为几乎完全双侧失明。两年后,她出现癫痫发作和认知障碍,最终诊断为NCL7,其病因是MFSD8基因的纯合突变。该病例强调了在以视锥视杆营养不良和视力丧失为主要临床特征的病例中,将NCLs视为潜在诊断的重要性。它还强调了与NCL7相关的视网膜变性在其他体征和症状出现之前的早发性和初始表现,这是该类情况的第二例有记录的病例。由于NCL7有可能在其他标志性体征出现之前最初表现为视力丧失,因此在鉴别诊断的各种综合征性和非综合征性疾病中考虑到它至关重要。