Dataset Information

Validation of the Italian version of the pediatric CMT quality of life outcome measure.

ABSTRACT: The pediatric Charcot-Marie-Tooth (CMT) specific quality of life (QOL) outcome measure (pCMT-QOL) is a recently developed and validated patient-reported measure of health QOL for children with CMT. The aim of this study was to provide and validate an Italian version of the pCMT-QOL. The original English version was translated and adapted into Italian using standard procedures. pCMT-QOL was administered to patients genetically diagnosed with CMT, aged 8 to 18 years. A retest was given 2 weeks later to assess reliability in all patients. A total of 22 patients (median age 14 years, DS 2.5; M:F 1:1) affected with CMT (19 CMT1A, 2 CMT2A, 1 CMT2K) were assessed as part of their clinical visit. The Italian-pCMT-QOL demonstrate a high test-retest reliability. None of the patients experienced difficulty in completing the questionnaire, no further corrections were needed after administration in patients. The Italian-pCMT-QOL is a reliable, culturally adapted and comparable version of the original English pCMT-QOL. This questionnaire is expected to be valuable in monitoring disease progression and useful for future clinical trials in Italian-speaking children with CMT.

SUBMITTER: Moroni I

PROVIDER: S-EPMC9324941 | biostudies-literature | 2022 Jun

REPOSITORIES: biostudies-literature

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Validation of the Italian version of the pediatric CMT quality of life outcome measure.

Moroni Isabella I Danti Federica Rachele FR Pareyson Davide D Pagliano Emanuela E Piscosquito Giuseppe G Foscan Maria M Marchi Alessia A Ardissone Anna A Genitrini Silvia S Wu Tong Tong TT Shy Michael E ME Ramchandren Sindhu S

Journal of the peripheral nervous system : JPNS 20220503 2

The pediatric Charcot-Marie-Tooth (CMT) specific quality of life (QOL) outcome measure (pCMT-QOL) is a recently developed and validated patient-reported measure of health QOL for children with CMT. The aim of this study was to provide and validate an Italian version of the pCMT-QOL. The original English version was translated and adapted into Italian using standard procedures. pCMT-QOL was administered to patients genetically diagnosed with CMT, aged 8 to 18 years. A retest was given 2 weeks lat ...[more]

PMID: 35416371

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Project description:BACKGROUND:The 12-item International Hip Outcome Tool (iHOT12) is a patient-reported outcome (PRO) designed to evaluate quality of life. We assessed the psychometric properties of the Greek version (iHOT12-Gr) in hip osteoarthritic patients. METHODS:Data from 124 patients aged > 50 years were used for factor analysis. Reliability evaluation included internal consistency, test-retest reliability, and interpretability. Content validity was examined by calculating the item-level content validity indices (I-CVI) and the scale-level content validity indices (S-CVI), using two methods: S-CVI Average (S-CVI/Ave), and the S-CVI Universal Agreement among experts (S-CVI/UA). Construct validity was tested against Greek versions of the Lower Extremity Functional Scale (LEFS-Greek), Modified Harris Hip Score (MHHS-Gr), and the 30 s chair-to-stand, Timed Up & Go (TUG), and 9-stairs-ascend/descend (9S-A/D) tests. Known-groups validity was examined using LEFS-Greek (cut-off = 53 points) as estimate variable. Responsiveness was examined pre and post total hip arthroplasty (4 and 8 weeks). RESULTS:Factor analysis revealed a two-factor model. Factor-1 (items 1-9) reflects "Symptoms and functionality", while Factor-2 (items 10-12) reflects "Hip disorder-related concerns". Reliability: Internal consistency and test-retest reliability of iHOT12-Gr-total were excellent: Cronbach's alpha > 0.92 and ICC(95% CI) > 0.976(0.96-0.99)(p < 0.001). Interpretability: There was no floor or ceiling effect; measurement error: 3.72 (Factor-1), 3.64 (Factor-2), and 3.22 (iHOT12-Gr-total); minimal detectable change: 10.3 (Factor-1), 10.1 (Factor-2), and 8.92 (iHOT12-Gr-total). VALIDITY:Content validity: The I-CVI value of the 12 items ranged from 1.00 to 0.83, the S-CVI/Ave was 0.97 and the S-CVI/UA was 0.83. Construct validity: iHOT12-Gr correlated strongly with both LEFS-Greek and MHHS-Gr, and weakly but significantly with 30s chair-to-stand, TUG and 9S-A/D (p < 0.001). Known-groups validity showed that iHOT12-Gr well discriminated subgroups of patients (p < 0.001). ROC analysis cut-off points were 51.9 (Factor-1), 25 (Factor-2) and 45.2 (iHOT12-Gr-total) (p < 0.001). Responsiveness: Four and 8 weeks postoperatively, standardized response means of Factor-1, Factor-2, and iHOT12-Gr-total were > 0.8. CONCLUSION:iHOT12-Gr showed excellent reliability properties. The content validity was excellent and significant weak-to-strong correlations were found regarding construct validity. The known-group validity was also significant, while the responsiveness was excellent. iHOT12-Gr could be a reliable and valid PRO for assessing quality of life in patients with hip osteoarthritis.

Project description:BackgroundThe Pediatric Quality of Life™ 3.0 Neuromuscular Module is an instrument to assess health-related quality of life (HRQoL) among children with neuromuscular diseases (NMDs) aged 2-18 years. This study aimed to determine whether the PedsQL™ 3.0 Neuromuscular Module Indonesian Version is valid and reliable.MethodsThis study used the Indonesian translation of the PedsQL™ 3.0 Neuromuscular Module after getting formal permission from the inventor, and the translation process followed the Mapi linguistic translation guidelines. This study administered the PedsQL™ 3.0 Neuromuscular Module Indonesian Version to 84 parents and 71 children. In addition, we used the Rasch model to analyze the psychometric properties.ResultsThe reliability of the total scale of the PedsQL™ Neuromuscular Module Indonesian Version shows good to very good criteria. On the parent proxy-report, Cronbach alpha was 0.95, person reliability was 0.84, item reliability was 0.93, person separation was 2.32, item separation was 3.61, person strata separation was 4 levels, and item strata separation was 5 levels. On the child self-report, Cronbach alpha was 0.93, person reliability was 0.81, item reliability was 0.81, person separation was 2.08, item separation was 2.06, person strata separation was 3 levels, and item strata separation was 3 levels. The total scale of the PedsQL Neuromuscular Module Indonesian Version shows fair to good construct validity in parent proxy-report (explained variance 51.9%; unexplained variance 8.4%) and child self-report (explained variance 40.9%; unexplained variance 12.6%). There were no misfit items in the parent proxy-report (infit 0.66-1.49; outfit 0.51-1.81; point measure correlation 0.36-0.93) and child self-report (infit 0.53-1.65; outfit 0.50-1.73; point measure correlation 0.31-0.90) identified by the Rasch models.ConclusionsThe PedsQL™ 3.0 Neuromuscular Modul Indonesian Version is a valid and reliable instrument in measuring HRQoL in Indonesian children with neuromuscular diseases.

Dataset Information

Validation of the Italian version of the pediatric CMT quality of life outcome measure.

Publications

Validation of the Italian version of the pediatric CMT quality of life outcome measure.

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