Autonomic dysfunction is a major feature of cerebellar ataxia, neuropathy, vestibular areflexia 'CANVAS' syndrome.

نویسندگان

  • Teddy Y Wu
  • Jennifer M Taylor
  • Dean H Kilfoyle
  • Andrew D Smith
  • Ben J McGuinness
  • Mark P Simpson
  • Elizabeth B Walker
  • Peter S Bergin
  • James C Cleland
  • David O Hutchinson
  • Neil E Anderson
  • Barry J Snow
  • Tim J Anderson
  • Laura A F Paermentier
  • Nick J Cutfield
  • Andrew M Chancellor
  • Stuart S Mossman
  • Richard H Roxburgh
چکیده

Cerebellar ataxia, neuropathy and vestibular areflexia syndrome (CANVAS) is a recently recognized neurodegenerative ganglionopathy. Prompted by the presence of symptomatic postural hypotension in two patients with CANVAS, we hypothesized that autonomic dysfunction may be an associated feature of the syndrome. We assessed symptoms of autonomic dysfunction and performed autonomic nervous system testing among 26 patients from New Zealand. After excluding three patients with diabetes mellitus, 83% had evidence of autonomic dysfunction; all patients had at least one autonomic symptom and 91% had more than two symptoms. We also found a higher rate of downbeat nystagmus (65%) than previously described in CANVAS. We confirmed that sensory findings on nerve conduction tests were consistent with a sensory ganglionopathy and describe two patients with loss of trigeminal sensation consistent with previous pathological descriptions of trigeminal sensory ganglionopathy. Our results suggest that autonomic dysfunction is a major feature of CANVAS. This has implications for the management of patients with CANVAS as the autonomic symptoms may be amenable to treatment. The findings also provide an important differential diagnosis from multiple system atrophy for patients who present with ataxia and autonomic failure.

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Neurophysiological evidence for generalized sensory neuronopathy in cerebellar ataxia with neuropathy and bilateral vestibular areflexia syndrome.

INTRODUCTION Cerebellar ataxia with neuropathy and bilateral vestibular areflexia syndrome (CANVAS) is a recently described multisystem ataxia defined by the presence of cerebellar ataxia, bilateral vestibulopathy, and a somatosensory deficit. The characteristic clinical sign is an abnormal visually enhanced vestibuloocular reflex. The somatosensory deficit contributes to a significant level of...

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SCIENTIFIC COMMENTARIES Autonomic failure in CANVAS syndrome

A series of papers published in the 1990s by the Bronstein group triggered—albeit unintentionally—intensified research into a clinical syndrome characterized by a combination of selective bilateral vestibulopathy and cerebellar ataxia (Bronstein et al., 1991; Waterston et al., 1992; Rinne et al., 1995). The term ‘cerebellar ataxia, neuropathy, vestibular areflexia syndrome’ (CANVAS) was subsequ...

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Op-brai140197 1..8

REPORT Autonomic dysfunction is a major feature of cerebellar ataxia, neuropathy, vestibular areflexia ‘CANVAS’ syndrome Teddy Y. Wu, Jennifer M. Taylor, Dean H. Kilfoyle, Andrew D. Smith, Ben J. McGuinness, Mark P. Simpson, Elizabeth B. Walker, Peter S. Bergin, James C. Cleland, David O. Hutchinson, Neil E. Anderson, Barry J. Snow, Tim J. Anderson, Laura A.F. Paermentier, Nick J. Cutfield, And...

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CANVAS an update: clinical presentation, investigation and management.

BACKGROUND Cerebellar Ataxia with Neuropathy and bilateral Vestibular Areflexia Syndrome (CANVAS) is a multi-system ataxia which results in cerebellar ataxia, a bilateral vestibulopathy and a somatosensory deficit. This sensory deficit has recently been shown to be a neuronopathy, with marked dorsal root ganglia neuronal loss. The characteristic oculomotor clinical sign is an abnormal visually ...

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A 75-year-old woman complained about insecure gait since age 55. Clinical examination revealed signs of cerebellar ataxia, bilateral vestibulopathy, and peripheral sensory impairment. Sensory nerve action potentials were absent. The visually enhanced vestibulo-ocular reflex (VVOR) was impaired (video on the Neurology(®) Web site at www.neurology.org, figure 1) and the diagnosis of cerebellar at...

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عنوان ژورنال:
  • Brain : a journal of neurology

دوره 137 Pt 10  شماره 

صفحات  -

تاریخ انتشار 2014