Reliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease.

Hdl Handle:
http://hdl.handle.net/10147/254534
Title:
Reliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease.
Authors:
Murphy, Sinéad M; Herrmann, David N; McDermott, Michael P; Scherer, Steven S; Shy, Michael E; Reilly, Mary M; Pareyson, Davide
Affiliation:
MRC Centre for Neuromuscular Diseases, National Hospital for Neurology and Neurosurgery, London, UK. sinead.murphy@uclh.nhs.uk
Citation:
Reliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease. 2011, 16 (3):191-8 J. Peripher. Nerv. Syst.
Publisher:
Journal of the peripheral nervous system : JPNS
Journal:
Journal of the peripheral nervous system : JPNS
Issue Date:
Sep-2011
URI:
http://hdl.handle.net/10147/254534
DOI:
10.1111/j.1529-8027.2011.00350.x
PubMed ID:
22003934
Abstract:
The 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.
Item Type:
Article
Language:
en
MeSH:
Adult; Charcot-Marie-Tooth Disease; Female; Humans; Male; Middle Aged; Neurologic Examination; Observer Variation; Reproducibility of Results; Sensitivity and Specificity; Young Adult
ISSN:
1529-8027

Full metadata record

DC FieldValue Language
dc.contributor.authorMurphy, Sinéad Men_GB
dc.contributor.authorHerrmann, David Nen_GB
dc.contributor.authorMcDermott, Michael Pen_GB
dc.contributor.authorScherer, Steven Sen_GB
dc.contributor.authorShy, Michael Een_GB
dc.contributor.authorReilly, Mary Men_GB
dc.contributor.authorPareyson, Davideen_GB
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.en_GB
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

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