Department of Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, UK.
Autonomic Unit, National Hospital for Neurology and Neurosurgery, Queen Square, London, UK.
J Peripher Nerv Syst. 2022 Dec;27(4):311-315. doi: 10.1111/jns.12515. Epub 2022 Oct 7.
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.
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。