RELATIONSHIP BETWEEN PROPRIOCEPTION AT THE KNEE JOINT AND GAIT ATAXIA IN HSAN III
Identifieur interne : 000328 ( Pmc/Curation ); précédent : 000327; suivant : 000329RELATIONSHIP BETWEEN PROPRIOCEPTION AT THE KNEE JOINT AND GAIT ATAXIA IN HSAN III
Auteurs : Vaughan G. Macefield [Australie] ; Lucy J. Norcliffe-Kaufmann [États-Unis] ; Felicia B. Axelrod [États-Unis] ; Horacio Kaufmann [États-Unis]Source :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2013.
Abstract
Hereditary sensory and autonomic neuropathy type III features a marked ataxic gait that progressively worsens over time. We assessed whether proprioceptive disturbances can explain the ataxia. Proprioception at the knee joint was assessed using passive joint angle matching in 18 patients and 14 age-matched controls; 5 patients with cerebellar ataxia were also studied. Ataxia was quantified using the Brief Ataxia Rating Score, which ranged from 7 to 26/30. Neuropathy patients performed poorly in judging joint position: mean absolute error was 8.7±1.0° and the range was very wide (2.8–18.1°); conversely, absolute error was only 2.7±0.3° (1.6–5.5°) in the controls and 3.0±0.2° (2.1–3.4°) in the cerebellar patients. This error was positively correlated to the degree of ataxia in the neuropathy patients but not the cerebellar patients, suggesting that poor proprioceptive acuity at the knee joint is a major contributor to the ataxic gait associated with hereditary sensory and autonomic neuropathy type III.
Url:
DOI: 10.1002/mds.25482
PubMed: 23681701
PubMed Central: 3694996
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Vaughan G. Macefield<affiliation><nlm:aff id="A2">School of Medicine, University of Western Sydney</nlm:aff>
<wicri:noCountry code="subfield">University of Western Sydney</wicri:noCountry>
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<country xml:lang="fr">Australie</country>
<wicri:regionArea>Neuroscience Research Australia, Sydney</wicri:regionArea>
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<front><div type="abstract" xml:lang="en"><p id="P1">Hereditary sensory and autonomic neuropathy type III features a marked ataxic gait that progressively worsens over time. We assessed whether proprioceptive disturbances can explain the ataxia. Proprioception at the knee joint was assessed using passive joint angle matching in 18 patients and 14 age-matched controls; 5 patients with cerebellar ataxia were also studied. Ataxia was quantified using the Brief Ataxia Rating Score, which ranged from 7 to 26/30. Neuropathy patients performed poorly in judging joint position: mean absolute error was 8.7±1.0° and the range was very wide (2.8–18.1°); conversely, absolute error was only 2.7±0.3° (1.6–5.5°) in the controls and 3.0±0.2° (2.1–3.4°) in the cerebellar patients. This error was positively correlated to the degree of ataxia in the neuropathy patients but not the cerebellar patients, suggesting that poor proprioceptive acuity at the knee joint is a major contributor to the ataxic gait associated with hereditary sensory and autonomic neuropathy type III.</p>
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<title-group><article-title>RELATIONSHIP BETWEEN PROPRIOCEPTION AT THE KNEE JOINT AND GAIT ATAXIA IN HSAN III</article-title>
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<contrib-group><contrib contrib-type="author"><name><surname>Macefield</surname>
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<aff id="A1"><label>1</label>
Dysautonomia Center, NYU Langone Medical Center, New York, USA</aff>
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School of Medicine, University of Western Sydney</aff>
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Neuroscience Research Australia, Sydney, Australia</aff>
<author-notes><corresp id="FN1">Corresponding author: Prof Vaughan Macefield, School of Medicine, University of Western Sydney, Locked Bag 1797, Penrith NSW 2751, Australia, Phone: +61 2 4620 3779, Fax: +61 2 4620 3890, <email>v.macefield@uws.edu.au</email>
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<pub-date pub-type="pmc-release"><day>01</day>
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<volume>28</volume>
<issue>6</issue>
<fpage>823</fpage>
<lpage>827</lpage>
<abstract><p id="P1">Hereditary sensory and autonomic neuropathy type III features a marked ataxic gait that progressively worsens over time. We assessed whether proprioceptive disturbances can explain the ataxia. Proprioception at the knee joint was assessed using passive joint angle matching in 18 patients and 14 age-matched controls; 5 patients with cerebellar ataxia were also studied. Ataxia was quantified using the Brief Ataxia Rating Score, which ranged from 7 to 26/30. Neuropathy patients performed poorly in judging joint position: mean absolute error was 8.7±1.0° and the range was very wide (2.8–18.1°); conversely, absolute error was only 2.7±0.3° (1.6–5.5°) in the controls and 3.0±0.2° (2.1–3.4°) in the cerebellar patients. This error was positively correlated to the degree of ataxia in the neuropathy patients but not the cerebellar patients, suggesting that poor proprioceptive acuity at the knee joint is a major contributor to the ataxic gait associated with hereditary sensory and autonomic neuropathy type III.</p>
</abstract>
<kwd-group><kwd>congenital insensitivity to pain</kwd>
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<kwd>hereditary sensory & autonomic neuropathy</kwd>
<kwd>muscle spindles</kwd>
<kwd>proprioception</kwd>
<kwd>Riley-Day syndrome</kwd>
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<funding-group><award-group><funding-source country="United States">National Institute of Neurological Disorders and Stroke : NINDS</funding-source>
<award-id>U54 NS065736 || NS</award-id>
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