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先天性肌无力综合征中反复出现的 COLQ 突变。

Recurrent COLQ mutation in congenital myasthenic syndrome.

机构信息

Department of Pediatric Neurology, Ankara Pediatric Disease Hematology and Oncology Training and Research Hospital, Ankara, Turkey.

出版信息

Pediatr Neurol. 2012 Apr;46(4):253-6. doi: 10.1016/j.pediatrneurol.2012.02.003.

Abstract

Congenital myasthenic syndromes comprise clinically and genetically heterogeneous disorders resulting from presynaptic, synaptic, or postsynaptic defects. Mutations in the COLQ gene result in acetylcholinesterase deficiency and cause a rare, autosomal recessive synaptic form of congenital myasthenic syndrome, with variable age of onset and clinical severity. We present four unrelated patients with a homozygous W148X mutation in the COLQ gene. Signs began at birth in all, but subsequent severity ranged from independent ambulation to wheelchair use during childhood. Treatment was partly effective; one patient was asymptomatic with 3,4-diaminopyridine treatment. These cases illustrate the clinical features and treatment results associated with this particular genotype, which appears to be relatively frequent among Turkish patients with congenital myasthenic syndrome.

摘要

先天性肌无力综合征包括由突触前、突触或突触后缺陷引起的具有临床和遗传异质性的疾病。COLQ 基因突变导致乙酰胆碱酯酶缺乏,并引起罕见的常染色体隐性突触性先天性肌无力综合征,发病年龄和临床严重程度存在差异。我们介绍了 4 例 COLQ 基因中纯合 W148X 突变的无关患者。所有患者的症状均在出生时出现,但随后的严重程度从儿童期独立行走到使用轮椅不等。治疗部分有效;1 名患者在使用 3,4-二氨基吡啶治疗后无症状。这些病例说明了与这种特定基因型相关的临床特征和治疗结果,这种基因型在土耳其先天性肌无力综合征患者中似乎较为常见。

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