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March 04, 2014; 82 (9) Article

Effect of pain in pediatric inherited neuropathies

Sindhu Ramchandren, Mamta Jaiswal, Eva Feldman, Michael Shy
First published January 29, 2014, DOI: https://doi.org/10.1212/WNL.0000000000000173
Sindhu Ramchandren
From the Department of Neurology (S.R., E.F.), University of Michigan Medical Health System, Ann Arbor, MI; and Department of Neurology (M.J., M.S.), University of Iowa, Iowa City.
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Mamta Jaiswal
From the Department of Neurology (S.R., E.F.), University of Michigan Medical Health System, Ann Arbor, MI; and Department of Neurology (M.J., M.S.), University of Iowa, Iowa City.
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Eva Feldman
From the Department of Neurology (S.R., E.F.), University of Michigan Medical Health System, Ann Arbor, MI; and Department of Neurology (M.J., M.S.), University of Iowa, Iowa City.
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Michael Shy
From the Department of Neurology (S.R., E.F.), University of Michigan Medical Health System, Ann Arbor, MI; and Department of Neurology (M.J., M.S.), University of Iowa, Iowa City.
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Citation
Effect of pain in pediatric inherited neuropathies
Sindhu Ramchandren, Mamta Jaiswal, Eva Feldman, Michael Shy
Neurology Mar 2014, 82 (9) 793-797; DOI: 10.1212/WNL.0000000000000173

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Abstract

Objective: Assess the prevalence and impact of pain in children with Charcot-Marie-Tooth (CMT) disease.

Methods: In this prospective cross-sectional study on children with CMT disease seen at study sites of the Inherited Neuropathy Consortium, we collected standardized assessments of pain (Wong-Baker FACES Pain Rating Scale) from 176 patients (140 children aged 8–18 years, and 36 children aged 2–7 years through parent proxies), along with standardized clinical assessments and quality-of-life (QOL) outcomes. We then developed a series of multivariate regression models to determine whether standardized measures of neuropathy severity, functional impact, or structural changes to the feet explained the observed pain scores.

Results: The mean score on the Wong-Baker FACES Pain Rating Scale was 2 (range 0–5). Increased pain strongly correlated with worse QOL scores but not with more severe neuropathy. Independent determinants of increased pain in children with CMT disease included measures of ankle inflexibility.

Conclusion: Pain is present in children with CMT disease and negatively affects QOL. Pain scores do not positively correlate with neuropathy severity but do correlate in limited univariate analyses with measures of ankle inflexibility. Further studies to elucidate the mechanisms of pain may help identify treatments that can reduce pain and improve QOL in patients with CMT disease.

GLOSSARY

CHQ=
Child Health Questionnaire;
CMT=
Charcot-Marie-Tooth;
CMTNSv2=
Charcot-Marie-Tooth Neuropathy Score version 2;
QOL=
quality of life

Footnotes

  • Go to Neurology.org for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Received August 6, 2013.
  • Accepted in final form November 15, 2013.
  • © 2014 American Academy of Neurology
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