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CADASIL 伴不典型临床表现、磁共振成像及新突变:两例病例报告及文献复习

CADASIL with Atypical Clinical Symptoms, Magnetic Resonance Imaging, and Novel Mutations: Two Case Reports and a Review of the Literature.

机构信息

Department of Neurology, Balıkesir University, 10300, Balıkesir, Turkey.

Department of Neurology, Celal Bayar University, 45000, Manisa, Turkey.

出版信息

J Mol Neurosci. 2019 Aug;68(4):529-538. doi: 10.1007/s12031-019-01313-z. Epub 2019 Apr 16.

DOI:10.1007/s12031-019-01313-z
PMID:30993645
Abstract

Cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL) is a hereditary microangiopathy with adult onset caused by a missense mutation in the NOTCH3 gene in chromosome 19p13. It presents with autosomal dominant arteriopathy, subcortical infarctions, and leukoencephalopathy. Its common clinical presentations are seen as recurrent strokes, migraine or migraine-like headaches, progressive dementia, pseudobulbar paralysis, and psychiatric conditions. Two patients with CADASIL syndrome, whose diagnosis was made based on clinical course, age of onset, imaging findings, and genetic assays in the patients and family members, are presented here because of new familial polymorphisms. The first patient, with cerebellar and psychotic findings, had widespread non-confluent hyperintense lesions as well as moderate cerebellar atrophy in cranial magnetic resonance scanning. The other patient, with headache, dizziness, and forgetfulness, had gliotic lesions in both cerebral hemispheres. CADASIL gene studies revealed a new polymorphism in exon 33 in the first patient. In the other patient, the NOTCH3 gene was identified as a new variant of p.H243P (c.728A > C heterozygous). By reporting a family presenting with various clinical symptoms in the presence of new polymorphisms, we emphasize that CADASIL syndrome may present with various clinical courses and should be considered in differential diagnoses.

摘要

伴有皮质下梗死和白质脑病的常染色体显性脑动脉病(CADASIL)是一种常染色体显性遗传性小血管病,由染色体 19p13 上 NOTCH3 基因的错义突变引起。其表现为常染色体显性脑动脉病、皮质下梗死和白质脑病。其常见的临床表现为复发性中风、偏头痛或偏头痛样头痛、进行性痴呆、假性延髓麻痹和精神疾病。这里报告了两例 CADASIL 综合征患者,他们的诊断是基于临床病程、发病年龄、影像学发现以及患者和家庭成员的基因检测。由于新的家族多态性,这两例患者的临床表现各不相同。第一个患者,有小脑和精神病学发现,头部磁共振扫描显示广泛非融合性高信号病变和中度小脑萎缩。另一个患者,有头痛、头晕和健忘,双侧大脑半球有胶质病变。CADASIL 基因研究发现第一个患者的外显子 33 中有一个新的多态性。在另一个患者中,NOTCH3 基因被鉴定为新的 p.H243P 变体(c.728A > C 杂合性)。通过报告一个家族中存在各种临床症状和新的多态性,我们强调 CADASIL 综合征可能表现出不同的临床病程,应在鉴别诊断中考虑。

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Chorea Associated with Genetic-Confirmed CADASIL.与基因确诊的大脑常染色体显性动脉病伴皮质下梗死和白质脑病(CADASIL)相关的舞蹈症
Mov Disord Clin Pract. 2023 Aug 8;10(9):1433-1435. doi: 10.1002/mdc3.13847. eCollection 2023 Sep.
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本文引用的文献

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Cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL) in two siblings with neuropsychiatric symptoms.两名有神经精神症状的同胞患伴有皮质下梗死和白质脑病的大脑常染色体显性动脉病(CADASIL)
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Two novel mutations and a previously unreported intronic polymorphism in the NOTCH3 gene.
单基因引起的中风。
Genes (Basel). 2021 Nov 23;12(12):1855. doi: 10.3390/genes12121855.
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Mutations in Gene may Promote the Clinical Presentation of Spinocerebellar Ataxia Type 37 Caused by Mutations in Gene.基因中的突变可能会促进由基因中的突变引起的37型脊髓小脑共济失调的临床表现。
Front Mol Biosci. 2021 Jun 16;8:668312. doi: 10.3389/fmolb.2021.668312. eCollection 2021.
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Ataxia Associated with CADASIL: a Pathology-Confirmed Case Report and Literature Review.伴皮质下梗死和白质脑病的脑动脉病(CADASIL)性共济失调:一例病理学确诊病例报告及文献复习。
Cerebellum. 2020 Dec;19(6):907-910. doi: 10.1007/s12311-020-01173-z.
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Prevalence and Atypical Clinical Characteristics of Mutations Among Patients Admitted for Acute Lacunar Infarctions.急性腔隙性脑梗死患者中突变的患病率及非典型临床特征
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CADASIL.CADASIL.
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Two Japanese CADASIL families exhibiting Notch3 mutation R75P not involving cysteine residue.两个日本的伴有皮质下梗死和白质脑病的常染色体显性遗传性脑动脉病(CADASIL)家系,表现出Notch3基因R75P突变,该突变不涉及半胱氨酸残基。
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