Quality of life, health-related quality of life, and associated factors in Huntington's disease : a systematic review
© 2023. The Author(s)..
BACKGROUND: Huntington's disease (HD) is a genetic, neurodegenerative disease. Due to the progressive nature of HD and the absence of a cure, (health-related) quality of life ((HR)QoL) is an important topic. Several studies have investigated (HR)QoL in HD, yet a clear synthesis of the existing literature is lacking to date. We performed a systematic review on self-reported (HR)QoL, and factors and intervention effects associated with (HR)QoL in premanifest and manifest HD gene expansion carriers (pHDGECs and mHDGECs, respectively).
METHODS: PubMed, EMBASE, Web of Science, and PsycINFO were searched systematically from September 17th, 2021, up to August 11th, 2022. Methodological and conceptual quality of the included studies was assessed with two appraisal tools.
RESULTS: 30 out of 70 eligible articles were included. mHDGECs experienced lower (HR)QoL compared to pHDGECs and controls, whereas mixed findings were reported when compared to other neurological diseases. Several factors were associated with (HR)QoL that might contribute to lower (HR)QoL in mHDGECs, including depressive symptoms, physical and psychological symptoms, lower functional capacity, lower support, and unmet needs. Multidisciplinary rehabilitation programs and a respiratory muscle training were beneficial for (HR)QoL in mHDGECs.
DISCUSSION: (HR)QoL is experienced differently across the course of the disease. Although (HR)QoL is key for understanding the impact of HD and the effect of symptomatic treatment, there is a need to improve the methodological and conceptual shortcomings that were found in most studies, especially regarding the conceptual clarity when reporting on QoL and HRQoL. Suggestions for strengthening these shortcomings are provided in this review.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2023 |
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| Erschienen: |
2023 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:270 |
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| Enthalten in: |
Journal of neurology - 270(2023), 5 vom: 30. Mai, Seite 2416-2437 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
van Lonkhuizen, Pearl J C [VerfasserIn] |
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| Links: |
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| Themen: |
Health-related quality of life |
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| Anmerkungen: |
Date Completed 27.04.2023 Date Revised 24.05.2023 published: Print-Electronic Citation Status MEDLINE |
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| doi: |
10.1007/s00415-022-11551-8 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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| PPN (Katalog-ID): |
NLM352261552 |
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| 100 | 1 | |a van Lonkhuizen, Pearl J C |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Quality of life, health-related quality of life, and associated factors in Huntington's disease |b a systematic review |
| 264 | 1 | |c 2023 | |
| 336 | |a Text |b txt |2 rdacontent | ||
| 337 | |a ƒaComputermedien |b c |2 rdamedia | ||
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| 500 | |a Date Completed 27.04.2023 | ||
| 500 | |a Date Revised 24.05.2023 | ||
| 500 | |a published: Print-Electronic | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © 2023. The Author(s). | ||
| 520 | |a BACKGROUND: Huntington's disease (HD) is a genetic, neurodegenerative disease. Due to the progressive nature of HD and the absence of a cure, (health-related) quality of life ((HR)QoL) is an important topic. Several studies have investigated (HR)QoL in HD, yet a clear synthesis of the existing literature is lacking to date. We performed a systematic review on self-reported (HR)QoL, and factors and intervention effects associated with (HR)QoL in premanifest and manifest HD gene expansion carriers (pHDGECs and mHDGECs, respectively) | ||
| 520 | |a METHODS: PubMed, EMBASE, Web of Science, and PsycINFO were searched systematically from September 17th, 2021, up to August 11th, 2022. Methodological and conceptual quality of the included studies was assessed with two appraisal tools | ||
| 520 | |a RESULTS: 30 out of 70 eligible articles were included. mHDGECs experienced lower (HR)QoL compared to pHDGECs and controls, whereas mixed findings were reported when compared to other neurological diseases. Several factors were associated with (HR)QoL that might contribute to lower (HR)QoL in mHDGECs, including depressive symptoms, physical and psychological symptoms, lower functional capacity, lower support, and unmet needs. Multidisciplinary rehabilitation programs and a respiratory muscle training were beneficial for (HR)QoL in mHDGECs | ||
| 520 | |a DISCUSSION: (HR)QoL is experienced differently across the course of the disease. Although (HR)QoL is key for understanding the impact of HD and the effect of symptomatic treatment, there is a need to improve the methodological and conceptual shortcomings that were found in most studies, especially regarding the conceptual clarity when reporting on QoL and HRQoL. Suggestions for strengthening these shortcomings are provided in this review | ||
| 650 | 4 | |a Systematic Review | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Review | |
| 650 | 4 | |a Health-related quality of life | |
| 650 | 4 | |a Huntington’s disease | |
| 650 | 4 | |a Patient-reported outcome measures | |
| 650 | 4 | |a Quality of life | |
| 650 | 4 | |a Systematic review | |
| 700 | 1 | |a Frank, Wiebke |e verfasserin |4 aut | |
| 700 | 1 | |a Heemskerk, Anne-Wil |e verfasserin |4 aut | |
| 700 | 1 | |a van Duijn, Erik |e verfasserin |4 aut | |
| 700 | 1 | |a de Bot, Susanne T |e verfasserin |4 aut | |
| 700 | 1 | |a Mühlbäck, Alzbeta |e verfasserin |4 aut | |
| 700 | 1 | |a Landwehrmeyer, G Bernhard |e verfasserin |4 aut | |
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| 700 | 1 | |a Chavannes, Niels H |e investigator |4 oth | |
| 700 | 1 | |a de Bot, Susanne T |e investigator |4 oth | |
| 700 | 1 | |a van Lonkhuizen, Pearl J C |e investigator |4 oth | |
| 700 | 1 | |a Landwehrmeyer, G Bernhard |e investigator |4 oth | |
| 700 | 1 | |a Steck, Franziska |e investigator |4 oth | |
| 700 | 1 | |a Klempíř, Jiří |e investigator |4 oth | |
| 700 | 1 | |a Konvalinková, Romama |e investigator |4 oth | |
| 700 | 1 | |a Bezuchová, Eva |e investigator |4 oth | |
| 700 | 1 | |a Dolečková, Kristýna |e investigator |4 oth | |
| 700 | 1 | |a Klempířová, Olga |e investigator |4 oth | |
| 700 | 1 | |a Roth, Jan |e investigator |4 oth | |
| 700 | 1 | |a Ulmanová, Olga |e investigator |4 oth | |
| 700 | 1 | |a Squitieri, Ferdinando |e investigator |4 oth | |
| 700 | 1 | |a Maffi, Sabrina |e investigator |4 oth | |
| 700 | 1 | |a Scaricamazza, Eugenia |e investigator |4 oth | |
| 700 | 1 | |a Migliore, Simone |e investigator |4 oth | |
| 700 | 1 | |a Di Giorgio, Chiara |e investigator |4 oth | |
| 700 | 1 | |a D'Alessio, Barbara |e investigator |4 oth | |
| 700 | 1 | |a Casella, Melissa |e investigator |4 oth | |
| 700 | 1 | |a Hoblyn, Jennifer |e investigator |4 oth | |
| 700 | 1 | |a Thangaramanujam, Muthukumaran |e investigator |4 oth | |
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| 700 | 1 | |a Kerr, Jacqueline |e investigator |4 oth | |
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