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dc.contributor.authorMurphy, Sinéad M
dc.contributor.authorHerrmann, David N
dc.contributor.authorMcDermott, Michael P
dc.contributor.authorScherer, Steven S
dc.contributor.authorShy, Michael E
dc.contributor.authorReilly, Mary M
dc.contributor.authorPareyson, Davide
dc.date.accessioned2012-12-05T10:12:12Z
dc.date.available2012-12-05T10:12:12Z
dc.date.issued2011-09
dc.identifier.citationReliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease. 2011, 16 (3):191-8 J. Peripher. Nerv. Syst.en_GB
dc.identifier.issn1529-8027
dc.identifier.pmid22003934
dc.identifier.doi10.1111/j.1529-8027.2011.00350.x
dc.identifier.urihttp://hdl.handle.net/10147/254534
dc.description.abstractThe Charcot-Marie-Tooth neuropathy score (CMTNS) is a reliable and valid composite score comprising symptoms, signs, and neurophysiological tests, which has been used in natural history studies of CMT1A and CMT1X and as an outcome measure in treatment trials of CMT1A. Following an international workshop on outcome measures in Charcot-Marie-Tooth disease (CMT), the CMTNS was modified to attempt to reduce floor and ceiling effects and to standardize patient assessment, aiming to improve its sensitivity for detecting change over time and the effect of an intervention. After agreeing on the modifications made to the CMTNS (CMTNS2), three examiners evaluated 16 patients to determine inter-rater reliability; one examiner evaluated 18 patients twice within 8 weeks to determine intra-rater reliability. Three examiners evaluated 63 patients using the CMTNS and the CMTNS2 to determine how the modifications altered scoring. For inter- and intra-rater reliability, intra-class correlation coefficients (ICCs) were ≥0.96 for the CMT symptom score and the CMT examination score. There were small but significant differences in some of the individual components of the CMTNS compared with the CMTNS2, mainly in the components that had been modified the most. A longitudinal study is in progress to determine whether the CMTNS2 is more sensitive than the CMTNS for detecting change over time.
dc.language.isoenen
dc.publisherJournal of the peripheral nervous system : JPNSen_GB
dc.rightsArchived with thanks to Journal of the peripheral nervous system : JPNSen_GB
dc.subject.meshAdult
dc.subject.meshCharcot-Marie-Tooth Disease
dc.subject.meshFemale
dc.subject.meshHumans
dc.subject.meshMale
dc.subject.meshMiddle Aged
dc.subject.meshNeurologic Examination
dc.subject.meshObserver Variation
dc.subject.meshReproducibility of Results
dc.subject.meshSensitivity and Specificity
dc.subject.meshYoung Adult
dc.titleReliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease.en_GB
dc.typeArticleen
dc.contributor.departmentMRC Centre for Neuromuscular Diseases, National Hospital for Neurology and Neurosurgery, London, UK. sinead.murphy@uclh.nhs.uken_GB
dc.identifier.journalJournal of the peripheral nervous system : JPNSen_GB
dc.description.provinceLeinsteren
html.description.abstractThe Charcot-Marie-Tooth neuropathy score (CMTNS) is a reliable and valid composite score comprising symptoms, signs, and neurophysiological tests, which has been used in natural history studies of CMT1A and CMT1X and as an outcome measure in treatment trials of CMT1A. Following an international workshop on outcome measures in Charcot-Marie-Tooth disease (CMT), the CMTNS was modified to attempt to reduce floor and ceiling effects and to standardize patient assessment, aiming to improve its sensitivity for detecting change over time and the effect of an intervention. After agreeing on the modifications made to the CMTNS (CMTNS2), three examiners evaluated 16 patients to determine inter-rater reliability; one examiner evaluated 18 patients twice within 8 weeks to determine intra-rater reliability. Three examiners evaluated 63 patients using the CMTNS and the CMTNS2 to determine how the modifications altered scoring. For inter- and intra-rater reliability, intra-class correlation coefficients (ICCs) were ≥0.96 for the CMT symptom score and the CMT examination score. There were small but significant differences in some of the individual components of the CMTNS compared with the CMTNS2, mainly in the components that had been modified the most. A longitudinal study is in progress to determine whether the CMTNS2 is more sensitive than the CMTNS for detecting change over time.


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