Sultan Çiçek, Miraç Yıldırım, Fatma Pınar Tabanlı, Engin Köse, Ömer Bektaş, Serap Teber
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引用次数: 0
摘要
10型神经蜡样脂肪褐变病(NCL10)是一种罕见的进行性神经退行性疾病,与编码组织蛋白酶D蛋白的CTSD基因纯合或复合杂合突变有关。根据出现症状的年龄分为先天性、婴儿期或青少年型NCL10。迄今为止,文献报道了6例青少年发病的NCL10 (JNCL10)。在此,我们报告了一个9岁6个月大的女孩,她患有语言障碍、认知和运动衰退、共济失调和视力障碍。据报道,7岁前的发育里程碑都是正常的。生化和代谢检查正常。脑电图显示轻度睡眠时间歇性广泛性高振幅δ波活动。脑磁共振成像显示轻度小脑萎缩。全外显子组测序(WES)在CTSD基因中发现了一个新的纯合错义变异c.1097G > a (p. Cys366Tyr)。根据临床、实验室和遗传学结果,该患者被诊断为JNCL10。据我们所知,这是一种新的变异,也是土耳其报道的第一例病例,在扩大种族和脑电图发现范围方面具有重要意义。
A Novel Variant of the CTSD Gene Associated with Juvenile-onset Neuronal Ceroid Lipofuscinosis Type 10: A Case Report and Literature Review.
Neuronal ceroid lipofuscinosis type 10 (NCL10) is a rare progressive neurodegenerative disease associated with homozygous or compound heterozygous mutations in the CTSD gene encoding cathepsin D protein. It is classified as congenital, infantile, or juvenile NCL10 according to the age at onset of symptoms. Six cases of juvenile onset NCL10 (JNCL10) have been reported thus far in the literature. Herein, we report a nine-year-six-month-old girl with speech disorders, cognitive and motor decline, ataxia, and visual impairment. Developmental milestones were reported to be normal up to the age of 7 years. Biochemical and metabolic studies were normal. Electroencephalography showed intermittent generalized high-amplitude delta-wave activity during light sleep. Brain magnetic resonance imaging showed mild cerebellar atrophy. Whole-exome sequencing (WES) revealed a novel homozygous missense variant of c.1097G > A (p. Cys366Tyr) in the CTSD gene. Based on clinical, laboratory, and genetic findings, the patient was diagnosed with JNCL10. To the best of our knowledge, this is a novel variant and the first case reported in Turkey, and it is important in terms of broadening ethnicity and the spectrum of EEG findings.
期刊介绍:
Official publication of the Society for Research on the Cerebellum devoted to genetics of cerebellar ataxias, role of cerebellum in motor control and cognitive function, and amid an ageing population, diseases associated with cerebellar dysfunction.
The Cerebellum is a central source for the latest developments in fundamental neurosciences including molecular and cellular biology; behavioural neurosciences and neurochemistry; genetics; fundamental and clinical neurophysiology; neurology and neuropathology; cognition and neuroimaging.
The Cerebellum benefits neuroscientists in molecular and cellular biology; neurophysiologists; researchers in neurotransmission; neurologists; radiologists; paediatricians; neuropsychologists; students of neurology and psychiatry and others.