Facioscapulohumeral muscular dystrophy Health Index : Japanese translation and validation study
PURPOSE: The Facioscapulohumeral Muscular Dystrophy Health Index (FSHD-HI) is a patient-reported outcome measure developed for patients with FSHD. This study aimed to translate the FSHD-HI into Japanese (FSHD-HI-J), evaluate cultural adaptation, and examine its psychometric properties.
MATERIALS AND METHODS: We created two forward translations, integrated them into a single Japanese version, and evaluated the back-translated version of the FSHD-HI. After finalizing the translation and cultural adaptation, we conducted a survey of 66 patients with FSHD to examine the reliability and validity of the FSHD-HI-J. For psychometric evaluations, we used Cronbach's alpha to assess internal consistency, the intraclass correlation coefficient (ICC) for test-retest reliability, and assessed validity based on the associations between FSHD-HI-J, clinical variables, and quality of life measures.
RESULTS: The FSHD-HI-J was found to be clinically relevant, indicating high internal consistency and test-retest reliability (ICC = 0.92 [95% confidence interval: 0.86-0.95] for the total score), as well as significant associations with clinical variables (D4Z4 repeats and functional impairment) and other quality of life measures (|rho| = 0.25-0.73).
CONCLUSIONS: The FSHD-HI-J is a valid and reliable patient-reported outcome measure for Japanese patients with FSHD. This validated, disease-specific patient-reported outcome is essential for future clinical practice and clinical trials.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2024 |
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| Erschienen: |
2024 |
| Enthalten in: |
Zur Gesamtaufnahme - year:2024 |
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| Enthalten in: |
Disability and rehabilitation - (2024) vom: 31. März, Seite 1-10 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Fujino, Haruo [VerfasserIn] |
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| Links: |
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| Themen: |
Disease burden |
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| Anmerkungen: |
Date Revised 31.03.2024 published: Print-Electronic Citation Status Publisher |
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| doi: |
10.1080/09638288.2024.2322035 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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| PPN (Katalog-ID): |
NLM370452569 |
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| 100 | 1 | |a Fujino, Haruo |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Facioscapulohumeral muscular dystrophy Health Index |b Japanese translation and validation study |
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| 500 | |a Date Revised 31.03.2024 | ||
| 500 | |a published: Print-Electronic | ||
| 500 | |a Citation Status Publisher | ||
| 520 | |a PURPOSE: The Facioscapulohumeral Muscular Dystrophy Health Index (FSHD-HI) is a patient-reported outcome measure developed for patients with FSHD. This study aimed to translate the FSHD-HI into Japanese (FSHD-HI-J), evaluate cultural adaptation, and examine its psychometric properties | ||
| 520 | |a MATERIALS AND METHODS: We created two forward translations, integrated them into a single Japanese version, and evaluated the back-translated version of the FSHD-HI. After finalizing the translation and cultural adaptation, we conducted a survey of 66 patients with FSHD to examine the reliability and validity of the FSHD-HI-J. For psychometric evaluations, we used Cronbach's alpha to assess internal consistency, the intraclass correlation coefficient (ICC) for test-retest reliability, and assessed validity based on the associations between FSHD-HI-J, clinical variables, and quality of life measures | ||
| 520 | |a RESULTS: The FSHD-HI-J was found to be clinically relevant, indicating high internal consistency and test-retest reliability (ICC = 0.92 [95% confidence interval: 0.86-0.95] for the total score), as well as significant associations with clinical variables (D4Z4 repeats and functional impairment) and other quality of life measures (|rho| = 0.25-0.73) | ||
| 520 | |a CONCLUSIONS: The FSHD-HI-J is a valid and reliable patient-reported outcome measure for Japanese patients with FSHD. This validated, disease-specific patient-reported outcome is essential for future clinical practice and clinical trials | ||
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Facioscapulohumeral muscular dystrophy | |
| 650 | 4 | |a disease burden | |
| 650 | 4 | |a neuromuscular disorder | |
| 650 | 4 | |a patient-reported outcomes | |
| 650 | 4 | |a quality of life | |
| 700 | 1 | |a Takahashi, Masanori P |e verfasserin |4 aut | |
| 700 | 1 | |a Nakamura, Harumasa |e verfasserin |4 aut | |
| 700 | 1 | |a Heatwole, Chad R |e verfasserin |4 aut | |
| 700 | 1 | |a Takada, Hiroto |e verfasserin |4 aut | |
| 700 | 1 | |a Kuru, Satoshi |e verfasserin |4 aut | |
| 700 | 1 | |a Ogata, Katsuhisa |e verfasserin |4 aut | |
| 700 | 1 | |a Enomoto, Kiyoka |e verfasserin |4 aut | |
| 700 | 1 | |a Hayashi, Yuto |e verfasserin |4 aut | |
| 700 | 1 | |a Imura, Osamu |e verfasserin |4 aut | |
| 700 | 1 | |a Matsumura, Tsuyoshi |e verfasserin |4 aut | |
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