Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility
Abstract Background Little is known about the spectrum of everyday challenges that people with skeletal dysplasia face because of their health and functioning. We aimed to identify factors related to health, functioning and disability in people with skeletal dysplasia, and their challenges with acce...
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| Format: | Article |
| Language: | English |
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BMC
2021-05-01
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| Series: | Orphanet Journal of Rare Diseases |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s13023-021-01857-7 |
| _version_ | 1831683000730910720 |
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| author | Heidi Anttila Susanna Tallqvist Minna Muñoz Sanna Leppäjoki-Tiistola Outi Mäkitie Sinikka Hiekkala |
| author_facet | Heidi Anttila Susanna Tallqvist Minna Muñoz Sanna Leppäjoki-Tiistola Outi Mäkitie Sinikka Hiekkala |
| author_sort | Heidi Anttila |
| collection | DOAJ |
| description | Abstract Background Little is known about the spectrum of everyday challenges that people with skeletal dysplasia face because of their health and functioning. We aimed to identify factors related to health, functioning and disability in people with skeletal dysplasia, and their challenges with accessibility in order to form a self-reported questionnaire for national data collection. The comprehensive musculoskeletal post-acute core set of the International Classification of Functioning, Disability and Health (ICF) was used as a framework. Methods An iterative, participatory and qualitative process was used to formulate a questionnaire. Items were searched from Patient-Reported Outcomes Measurement Information System and from other self-report instruments, additional items were formulated using ICF linking rules. Expert panels from the target population assessed the face and content validity in thematic interviews. Results The questionnaire demonstrated its relevance, comprehensiveness and feasibility for people with skeletal dysplasia. The ICF linkages showed the contents’ correspondence to the construct. Expert panels added 15 categories and one on chapter level to the core set and confirmed content validity. The final survey covers 86 ICF categories and 173 ICF-linked items that were grouped to 33 questions. Conclusions The content of the questionnaire proved to be sufficiently valid for people with skeletal dysplasia. It can be used to explore their health, functioning, disability and accessibility to develop care and rehabilitation policies, to plan services and to provide information to various parties involved. |
| first_indexed | 2024-12-20T06:28:28Z |
| format | Article |
| id | doaj.art-97d2a856896948b9acd22983e689f6c3 |
| institution | Directory Open Access Journal |
| issn | 1750-1172 |
| language | English |
| last_indexed | 2024-12-20T06:28:28Z |
| publishDate | 2021-05-01 |
| publisher | BMC |
| record_format | Article |
| series | Orphanet Journal of Rare Diseases |
| spelling | doaj.art-97d2a856896948b9acd22983e689f6c32022-12-21T19:50:13ZengBMCOrphanet Journal of Rare Diseases1750-11722021-05-0116111110.1186/s13023-021-01857-7Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibilityHeidi Anttila0Susanna Tallqvist1Minna Muñoz2Sanna Leppäjoki-Tiistola3Outi Mäkitie4Sinikka Hiekkala5Public Health and Welfare Department, Knowledge Management and Co-Creation Unit, Finnish Institute for Health and WelfareUniversity of HelsinkiValidia Ltd, Validia RehabilitationLyhytkasvuiset – Kortväxta ry (Finnish Association for People with Restricted Growth and for Their Families)Children’s Hospital, University of Helsinki and Helsinki University HospitalValidia Ltd, Validia RehabilitationAbstract Background Little is known about the spectrum of everyday challenges that people with skeletal dysplasia face because of their health and functioning. We aimed to identify factors related to health, functioning and disability in people with skeletal dysplasia, and their challenges with accessibility in order to form a self-reported questionnaire for national data collection. The comprehensive musculoskeletal post-acute core set of the International Classification of Functioning, Disability and Health (ICF) was used as a framework. Methods An iterative, participatory and qualitative process was used to formulate a questionnaire. Items were searched from Patient-Reported Outcomes Measurement Information System and from other self-report instruments, additional items were formulated using ICF linking rules. Expert panels from the target population assessed the face and content validity in thematic interviews. Results The questionnaire demonstrated its relevance, comprehensiveness and feasibility for people with skeletal dysplasia. The ICF linkages showed the contents’ correspondence to the construct. Expert panels added 15 categories and one on chapter level to the core set and confirmed content validity. The final survey covers 86 ICF categories and 173 ICF-linked items that were grouped to 33 questions. Conclusions The content of the questionnaire proved to be sufficiently valid for people with skeletal dysplasia. It can be used to explore their health, functioning, disability and accessibility to develop care and rehabilitation policies, to plan services and to provide information to various parties involved.https://doi.org/10.1186/s13023-021-01857-7FunctioningDisabilityEnvironmental factorsSelf-reportQuestionnaire designContent validity |
| spellingShingle | Heidi Anttila Susanna Tallqvist Minna Muñoz Sanna Leppäjoki-Tiistola Outi Mäkitie Sinikka Hiekkala Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility Orphanet Journal of Rare Diseases Functioning Disability Environmental factors Self-report Questionnaire design Content validity |
| title | Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility |
| title_full | Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility |
| title_fullStr | Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility |
| title_full_unstemmed | Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility |
| title_short | Towards an ICF-based self-report questionnaire for people with skeletal dysplasia to study health, functioning, disability and accessibility |
| title_sort | towards an icf based self report questionnaire for people with skeletal dysplasia to study health functioning disability and accessibility |
| topic | Functioning Disability Environmental factors Self-report Questionnaire design Content validity |
| url | https://doi.org/10.1186/s13023-021-01857-7 |
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