Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review
Introduction Bleeding disorders (BDs) may influence health‐related quality of life (HRQoL) in children and caregivers. Measuring HRQoL gives insight into domains requiring support and provides an opportunity to evaluate the effects of novel therapies. Aim To gain insight in the current body of liter...
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| Veröffentlicht in: | Haemophilia : the official journal of the World Federation of Hemophilia 2024-03, Vol.30 (2), p.295-305 |
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| container_title | Haemophilia : the official journal of the World Federation of Hemophilia |
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| creator | Huisman, Elise J. Mussert, Caroline Bai, Guannan Raat, Hein Cnossen, Marjon H. |
| description | Introduction
Bleeding disorders (BDs) may influence health‐related quality of life (HRQoL) in children and caregivers. Measuring HRQoL gives insight into domains requiring support and provides an opportunity to evaluate the effects of novel therapies.
Aim
To gain insight in the current body of literature on HRQoL in children with BDs in order to identify knowledge gaps for research and further development of this field.
Methods
Scoping review.
Results
We included 53 articles, describing studies mainly performed in Europe and North–America (60.4%) and mostly within the last ten years. Only 32% studies included children |
| doi_str_mv | 10.1111/hae.14941 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2922949909</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2955346704</sourcerecordid><originalsourceid>FETCH-LOGICAL-c3481-5fd6d5c9ec5c0b4c9076920f10cf64ef62c8b4a424cb0724c1f6a46f496800f83</originalsourceid><addsrcrecordid>eNp10cFOHCEcBnBianSrHvoCDUkv9jAKDMMOx43Z1qYmXvQ8YZg_Oxh2mIUZN3sx-whN-oY-iayrHpqUAxDy4wvhQ-gLJRc0jctWwQXlktMDNKG5KDJWUPFpty9oVjIqjtHnGB8IoTkj4ggd52VOpzznE_T0u_NrB80C8EL1EdsOt6Dc0D5v_wRwaoAGr0bl7LDB3mBnDeAAEVTQLe4hGB-WiaRrurWuCdDhtR1aXDuAxnYL3NjoQwMh4uftXzzDUft-dx7g0cL6FB0a5SKcva0n6P7H_O7qOru5_fnranaT6ZyXNCtMI5pCS9CFJjXXkkyFZMRQoo3gYATTZc0VZ1zXZJpmaoTiwnApSkJMmZ-g831uH_xqhDhUSxs1OKc68GOsmGRMcimJTPTbP_TBj6FLr0uqKHIupoQn9X2vdPAxBjBVH-xShU1FSbUrpUqlVK-lJPv1LXGs02d9yPcWErjcg7V1sPl_UnU9m-8jXwAgQZg9</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2955346704</pqid></control><display><type>article</type><title>Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review</title><source>Access via Wiley Online Library</source><creator>Huisman, Elise J. ; Mussert, Caroline ; Bai, Guannan ; Raat, Hein ; Cnossen, Marjon H.</creator><creatorcontrib>Huisman, Elise J. ; Mussert, Caroline ; Bai, Guannan ; Raat, Hein ; Cnossen, Marjon H.</creatorcontrib><description>Introduction
Bleeding disorders (BDs) may influence health‐related quality of life (HRQoL) in children and caregivers. Measuring HRQoL gives insight into domains requiring support and provides an opportunity to evaluate the effects of novel therapies.
Aim
To gain insight in the current body of literature on HRQoL in children with BDs in order to identify knowledge gaps for research and further development of this field.
Methods
Scoping review.
Results
We included 53 articles, describing studies mainly performed in Europe and North–America (60.4%) and mostly within the last ten years. Only 32% studies included children <4 years. Almost all studies (47/53, 88.7%) were performed in boys with haemophilia, pooling haemophilia A and B (n = 21) and different disease severities (n = 20). Thirteen different generic and five disease‐specific HRQoL‐questionnaires were applied; all questionnaires were validated for haemophilia specifically. Six (11,3%) combined generic and disease‐specific questionnaires. Self‐reports were most frequently applied (40/53, 75.5%), sometimes combined with proxy and/or parent‐reports (17/53, 32.1%). Eleven studies used a reference group (20.8%). Statistical analyses mostly consisted of mean and SD (77.4%).
Conclusion
HRQoL‐research is mainly performed in school‐aged boys with haemophilia, treated in developed countries. Pitfalls encountered are the pooling of various BDs, subtypes and severities, as well as the application of multiple generic questionnaires prohibiting comparison of results. More attention is needed for broader study populations including other BDs, young children, feminine bleeding issues and platelet disorders, as well as the use of HRQoL as an effect‐measurement tool for medical interventions, and more thorough statistical analysis.</description><identifier>ISSN: 1351-8216</identifier><identifier>EISSN: 1365-2516</identifier><identifier>DOI: 10.1111/hae.14941</identifier><identifier>PMID: 38317434</identifier><language>eng</language><publisher>England: Wiley Subscription Services, Inc</publisher><subject>Bleeding ; blood coagulation disorders [MESH] ; child [MESH] ; Children ; haemophilia [MESH] ; Hemophilia ; Population studies ; Quality of life ; quality of life [MESH] ; Questionnaires ; research design [MESH] ; review [MESH] ; Statistical analysis ; Statistics</subject><ispartof>Haemophilia : the official journal of the World Federation of Hemophilia, 2024-03, Vol.30 (2), p.295-305</ispartof><rights>2024 The Authors. published by John Wiley & Sons Ltd.</rights><rights>2024 The Authors. Haemophilia published by John Wiley & Sons Ltd.</rights><rights>2024. This article is published under http://creativecommons.org/licenses/by-nc-nd/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c3481-5fd6d5c9ec5c0b4c9076920f10cf64ef62c8b4a424cb0724c1f6a46f496800f83</cites><orcidid>0000-0003-1557-2995 ; 0000-0002-6000-7445 ; 0000-0003-0619-7580 ; 0000-0002-0700-2194</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fhae.14941$$EPDF$$P50$$Gwiley$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fhae.14941$$EHTML$$P50$$Gwiley$$Hfree_for_read</linktohtml><link.rule.ids>314,780,784,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/38317434$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Huisman, Elise J.</creatorcontrib><creatorcontrib>Mussert, Caroline</creatorcontrib><creatorcontrib>Bai, Guannan</creatorcontrib><creatorcontrib>Raat, Hein</creatorcontrib><creatorcontrib>Cnossen, Marjon H.</creatorcontrib><title>Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review</title><title>Haemophilia : the official journal of the World Federation of Hemophilia</title><addtitle>Haemophilia</addtitle><description>Introduction
Bleeding disorders (BDs) may influence health‐related quality of life (HRQoL) in children and caregivers. Measuring HRQoL gives insight into domains requiring support and provides an opportunity to evaluate the effects of novel therapies.
Aim
To gain insight in the current body of literature on HRQoL in children with BDs in order to identify knowledge gaps for research and further development of this field.
Methods
Scoping review.
Results
We included 53 articles, describing studies mainly performed in Europe and North–America (60.4%) and mostly within the last ten years. Only 32% studies included children <4 years. Almost all studies (47/53, 88.7%) were performed in boys with haemophilia, pooling haemophilia A and B (n = 21) and different disease severities (n = 20). Thirteen different generic and five disease‐specific HRQoL‐questionnaires were applied; all questionnaires were validated for haemophilia specifically. Six (11,3%) combined generic and disease‐specific questionnaires. Self‐reports were most frequently applied (40/53, 75.5%), sometimes combined with proxy and/or parent‐reports (17/53, 32.1%). Eleven studies used a reference group (20.8%). Statistical analyses mostly consisted of mean and SD (77.4%).
Conclusion
HRQoL‐research is mainly performed in school‐aged boys with haemophilia, treated in developed countries. Pitfalls encountered are the pooling of various BDs, subtypes and severities, as well as the application of multiple generic questionnaires prohibiting comparison of results. More attention is needed for broader study populations including other BDs, young children, feminine bleeding issues and platelet disorders, as well as the use of HRQoL as an effect‐measurement tool for medical interventions, and more thorough statistical analysis.</description><subject>Bleeding</subject><subject>blood coagulation disorders [MESH]</subject><subject>child [MESH]</subject><subject>Children</subject><subject>haemophilia [MESH]</subject><subject>Hemophilia</subject><subject>Population studies</subject><subject>Quality of life</subject><subject>quality of life [MESH]</subject><subject>Questionnaires</subject><subject>research design [MESH]</subject><subject>review [MESH]</subject><subject>Statistical analysis</subject><subject>Statistics</subject><issn>1351-8216</issn><issn>1365-2516</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><sourceid>WIN</sourceid><recordid>eNp10cFOHCEcBnBianSrHvoCDUkv9jAKDMMOx43Z1qYmXvQ8YZg_Oxh2mIUZN3sx-whN-oY-iayrHpqUAxDy4wvhQ-gLJRc0jctWwQXlktMDNKG5KDJWUPFpty9oVjIqjtHnGB8IoTkj4ggd52VOpzznE_T0u_NrB80C8EL1EdsOt6Dc0D5v_wRwaoAGr0bl7LDB3mBnDeAAEVTQLe4hGB-WiaRrurWuCdDhtR1aXDuAxnYL3NjoQwMh4uftXzzDUft-dx7g0cL6FB0a5SKcva0n6P7H_O7qOru5_fnranaT6ZyXNCtMI5pCS9CFJjXXkkyFZMRQoo3gYATTZc0VZ1zXZJpmaoTiwnApSkJMmZ-g831uH_xqhDhUSxs1OKc68GOsmGRMcimJTPTbP_TBj6FLr0uqKHIupoQn9X2vdPAxBjBVH-xShU1FSbUrpUqlVK-lJPv1LXGs02d9yPcWErjcg7V1sPl_UnU9m-8jXwAgQZg9</recordid><startdate>202403</startdate><enddate>202403</enddate><creator>Huisman, Elise J.</creator><creator>Mussert, Caroline</creator><creator>Bai, Guannan</creator><creator>Raat, Hein</creator><creator>Cnossen, Marjon H.</creator><general>Wiley Subscription Services, Inc</general><scope>24P</scope><scope>WIN</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>8FD</scope><scope>FR3</scope><scope>H94</scope><scope>P64</scope><scope>RC3</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0003-1557-2995</orcidid><orcidid>https://orcid.org/0000-0002-6000-7445</orcidid><orcidid>https://orcid.org/0000-0003-0619-7580</orcidid><orcidid>https://orcid.org/0000-0002-0700-2194</orcidid></search><sort><creationdate>202403</creationdate><title>Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review</title><author>Huisman, Elise J. ; Mussert, Caroline ; Bai, Guannan ; Raat, Hein ; Cnossen, Marjon H.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3481-5fd6d5c9ec5c0b4c9076920f10cf64ef62c8b4a424cb0724c1f6a46f496800f83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Bleeding</topic><topic>blood coagulation disorders [MESH]</topic><topic>child [MESH]</topic><topic>Children</topic><topic>haemophilia [MESH]</topic><topic>Hemophilia</topic><topic>Population studies</topic><topic>Quality of life</topic><topic>quality of life [MESH]</topic><topic>Questionnaires</topic><topic>research design [MESH]</topic><topic>review [MESH]</topic><topic>Statistical analysis</topic><topic>Statistics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Huisman, Elise J.</creatorcontrib><creatorcontrib>Mussert, Caroline</creatorcontrib><creatorcontrib>Bai, Guannan</creatorcontrib><creatorcontrib>Raat, Hein</creatorcontrib><creatorcontrib>Cnossen, Marjon H.</creatorcontrib><collection>Wiley Online Library Open Access</collection><collection>Wiley Online Library (Open Access Collection)</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Huisman, Elise J.</au><au>Mussert, Caroline</au><au>Bai, Guannan</au><au>Raat, Hein</au><au>Cnossen, Marjon H.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review</atitle><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle><addtitle>Haemophilia</addtitle><date>2024-03</date><risdate>2024</risdate><volume>30</volume><issue>2</issue><spage>295</spage><epage>305</epage><pages>295-305</pages><issn>1351-8216</issn><eissn>1365-2516</eissn><abstract>Introduction
Bleeding disorders (BDs) may influence health‐related quality of life (HRQoL) in children and caregivers. Measuring HRQoL gives insight into domains requiring support and provides an opportunity to evaluate the effects of novel therapies.
Aim
To gain insight in the current body of literature on HRQoL in children with BDs in order to identify knowledge gaps for research and further development of this field.
Methods
Scoping review.
Results
We included 53 articles, describing studies mainly performed in Europe and North–America (60.4%) and mostly within the last ten years. Only 32% studies included children <4 years. Almost all studies (47/53, 88.7%) were performed in boys with haemophilia, pooling haemophilia A and B (n = 21) and different disease severities (n = 20). Thirteen different generic and five disease‐specific HRQoL‐questionnaires were applied; all questionnaires were validated for haemophilia specifically. Six (11,3%) combined generic and disease‐specific questionnaires. Self‐reports were most frequently applied (40/53, 75.5%), sometimes combined with proxy and/or parent‐reports (17/53, 32.1%). Eleven studies used a reference group (20.8%). Statistical analyses mostly consisted of mean and SD (77.4%).
Conclusion
HRQoL‐research is mainly performed in school‐aged boys with haemophilia, treated in developed countries. Pitfalls encountered are the pooling of various BDs, subtypes and severities, as well as the application of multiple generic questionnaires prohibiting comparison of results. More attention is needed for broader study populations including other BDs, young children, feminine bleeding issues and platelet disorders, as well as the use of HRQoL as an effect‐measurement tool for medical interventions, and more thorough statistical analysis.</abstract><cop>England</cop><pub>Wiley Subscription Services, Inc</pub><pmid>38317434</pmid><doi>10.1111/hae.14941</doi><tpages>11</tpages><orcidid>https://orcid.org/0000-0003-1557-2995</orcidid><orcidid>https://orcid.org/0000-0002-6000-7445</orcidid><orcidid>https://orcid.org/0000-0003-0619-7580</orcidid><orcidid>https://orcid.org/0000-0002-0700-2194</orcidid><oa>free_for_read</oa></addata></record> |
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| ispartof | Haemophilia : the official journal of the World Federation of Hemophilia, 2024-03, Vol.30 (2), p.295-305 |
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| language | eng |
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| source | Access via Wiley Online Library |
| subjects | Bleeding blood coagulation disorders [MESH] child [MESH] Children haemophilia [MESH] Hemophilia Population studies Quality of life quality of life [MESH] Questionnaires research design [MESH] review [MESH] Statistical analysis Statistics |
| title | Knowledge gaps in health‐related quality of life research performed in children with bleeding disorders – A scoping review |
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