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DOI 10.1007/s12311-015-0684-6

Медицинская библиотека · Cerebellum (London, England) · 2016

Peripheral Neuropathy in Spinocerebellar Ataxia Type 1, 2, 3, and 6.

Linnemann Christoph, Tezenas du Montcel Sophie, Rakowicz Maryla, Schmitz-Hübsch Tanja, Szymanski Sandra, Berciano Jose, van de Warrenburg Bart P, Pedersen Karine, Depondt Chantal, Rola Rafal, Klockgether Thomas, García Antonio, Mutlu Gurkan, Schöls Ludger

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Spinocerebellar ataxias (SCAs) are characterized by autosomal dominantly inherited progressive ataxia but are clinically heterogeneous due to variable involvement of non-cerebellar parts of the nervous system. Non-cerebellar symptoms contribute significantly to the burden of SCAs, may guide the clinician to the underlying genetic subtype, and might be useful markers to monitor disease. Peripheral neuropathy is frequently observed in SCA, but subtype-specific features and subclinical manifestations have rarely been evaluated. We performed a multicenter nerve conduction study with 162 patients with genetically confirmed SCA1, SCA2, SCA3, and SCA6. The study proved peripheral nerves to be involved in the neurodegenerative process in 82 % of SCA1, 63 % of SCA2, 55 % of SCA3, and 22 % of SCA6 patients. Most patients of all subtypes revealed affection of both sensory and motor fibers. Neuropathy was most frequently of mixed type with axonal and demyelinating characteristics in all SCA subtypes. However, nerve conduction velocities of SCA1 patients were slower compared to other genotypes. SCA6 patients revealed less axonal damage than patients with other subtypes. No influence of CAG repeat length or biometric determinants on peripheral neuropathy could be identified in SCA1, SCA3, and SCA6. In SCA2, earlier onset and more severe ataxia were associated with peripheral neuropathy. We proved peripheral neuropathy to be a frequent site of the neurodegenerative process in all common SCA subtypes. Since damage to peripheral nerves is readily assessable by electrophysiological means, nerve conduction studies should be performed in a longitudinal approach to assess these parameters as potential progression markers.

Для цитирования: Linnemann Christoph, Tezenas du Montcel Sophie, Rakowicz Maryla, Schmitz-Hübsch Tanja, Szymanski Sandra, Berciano Jose, van de Warrenburg Bart P, Pedersen Karine, Depondt Chantal, Rola Rafal, Klockgether Thomas, García Antonio, Mutlu Gurkan, Schöls Ludger. Peripheral Neuropathy in Spinocerebellar Ataxia Type 1, 2, 3, and 6.. Cerebellum (London, England). 2016. DOI: 10.1007/s12311-015-0684-6.

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