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RFC1 相关疾病伴帕金森病严重自主神经功能障碍。

Severe distinct dysautonomia in RFC1-related disease associated with Parkinsonism.

机构信息

Department of Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.

Autonomic Unit, National Hospital for Neurology and Neurosurgery, Queen Square, London, UK.

出版信息

J Peripher Nerv Syst. 2022 Dec;27(4):311-315. doi: 10.1111/jns.12515. Epub 2022 Oct 7.

Abstract

Biallelic repeat expansions in replication factor C subunit 1 (RFC1) have recently been found to cause cerebellar ataxia, neuropathy and vestibular areflexia syndrome (CANVAS). Additional features that have been described include Parkinsonism and a multiple system atrophy (MSA)-like syndrome. CANVAS can include features of dysautonomia, but they are much milder than typically seen in MSA. We report a detailed autonomic phenotype of multisystem RFC1-related disease presenting initially as CANVAS. Our patient presented aged 61 with a sensory ataxic neuropathy who rapidly developed widespread autonomic failure and Parkinsonism. The autonomic profile was of a mixed pre- and post-ganglionic syndrome with progressive involvement of sympathetic and parasympathetic cardiovascular and sudomotor function. The Parkinsonism did not respond to levodopa. We present a patient with CANVAS and biallelic RFC1 expansions who developed Parkinsonism with severe autonomic involvement similar to that seen in classical MSA. The link between MSA and CANVAS remains uncertain.

摘要

最近发现复制因子 C 亚基 1(RFC1)的双等位基因重复扩展会导致小脑共济失调、神经病和前庭反射消失综合征(CANVAS)。此外还描述了其他特征,包括帕金森病和类似于多系统萎缩(MSA)的综合征。CANVAS 可能包括自主神经功能紊乱的特征,但比 MSA 中通常见到的要轻得多。我们报告了一个多系统 RFC1 相关疾病的详细自主神经表型,最初表现为 CANVAS。我们的患者 61 岁时出现感觉性共济失调性神经病,随后迅速出现广泛的自主神经衰竭和帕金森病。自主神经特征为混合节前和节后神经节综合征,伴有交感和副交感心血管和出汗功能的进行性受累。帕金森病对左旋多巴没有反应。我们报告了一名患有 CANVAS 和双等位基因 RFC1 扩展的患者,他出现了帕金森病和严重的自主神经受累,类似于经典 MSA 中见到的情况。MSA 和 CANVAS 之间的联系仍然不确定。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7bd3/10092280/dc0ecb15ceaa/JNS-27-311-g001.jpg

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