Impact on the family of a child’s rare disease: A large cross-sectional study
Introduction: Children living with rare diseases (RD) require considerable support from their families. Aim: To assess the impact of children’s RD on family functioning and evaluate predictors of the level of impact. Methods: Families of Australian children aged < 19 years and living with a RD we...
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| Language: | English |
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Elsevier
2025-01-01
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| Series: | Rare |
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| author | Judith M. Jobling Suzy M. Teutsch Guy D. Eslick Elizabeth J. Elliott |
| author_facet | Judith M. Jobling Suzy M. Teutsch Guy D. Eslick Elizabeth J. Elliott |
| author_sort | Judith M. Jobling |
| collection | DOAJ |
| description | Introduction: Children living with rare diseases (RD) require considerable support from their families. Aim: To assess the impact of children’s RD on family functioning and evaluate predictors of the level of impact. Methods: Families of Australian children aged < 19 years and living with a RD were invited to complete a comprehensive survey. Impact on family (IOF) score, using the validated IOF scale, embedded within the survey, was used as the main outcome measure. Results: Families of 462 children with > 240 different RD completed the survey. IOF scores increased as child health functioning (HF) worsened (Odds ratio [OR]= 97.3 [95 % CI 40.6–233.4] for severely reduced compared to excellent HF). Speaking a first language other than English (OR 2.9; 95 % CI 1.4–6.3), experiencing financial difficulty (OR 2.9; 95 % CI 1.4–3.1), long wait times to see a specialist doctor (OR 1.9; 95 % CI 1.3–2.9), and having two versus one child with a RD (OR 3.8; 95 % CI 1.1–14.5) were also associated with higher IOF scores. Conversely, IOF scores significantly decreased as the age of children increased (6–11 y: OR 0.6 (95 % CI 0.4–0.9); 12–18 y: OR 0.5 (95 % CI 0.3–0.8), versus 0–5 y). Conclusion: Worse health-related functioning in children with RD results in significantly higher impacts on family functioning. Practical, financial, and socio-emotional support is required to reduce the burden for families whose children live with a RD, particularly families from non-English speaking backgrounds, with financial stress, or with young children who have severe disease. |
| format | Article |
| id | doaj-art-3f39cfc071d8490e92abfdc0e0f04e02 |
| institution | DOAJ |
| issn | 2950-0087 |
| language | English |
| publishDate | 2025-01-01 |
| publisher | Elsevier |
| record_format | Article |
| series | Rare |
| spelling | doaj-art-3f39cfc071d8490e92abfdc0e0f04e022025-08-20T03:11:47ZengElsevierRare2950-00872025-01-01310006610.1016/j.rare.2025.100066Impact on the family of a child’s rare disease: A large cross-sectional studyJudith M. Jobling0Suzy M. Teutsch1Guy D. Eslick2Elizabeth J. Elliott3The University of Sydney, Faculty of Medicine and Health, Sydney Medical School, Sydney, New South Wales 2006, Australia; The University of Sydney, Faculty of Medicine and Health, Specialty of Child and Adolescent Health, Westmead, New South Wales 2145, Australia; The Australian Paediatric Surveillance Unit, Kids Research, Sydney Children’s Hospitals Network, Westmead, New South Wales 2145, AustraliaThe University of Sydney, Faculty of Medicine and Health, Specialty of Child and Adolescent Health, Westmead, New South Wales 2145, Australia; The Australian Paediatric Surveillance Unit, Kids Research, Sydney Children’s Hospitals Network, Westmead, New South Wales 2145, Australia; Correspondence to: The Australian Paediatric Surveillance Unit, Kids Research Sydney Children’s Hospitals Network, 214 Hawkesbury Road, Westmead, New South Wales 2145, Australia.The University of Sydney, Faculty of Medicine and Health, Specialty of Child and Adolescent Health, Westmead, New South Wales 2145, Australia; The Australian Paediatric Surveillance Unit, Kids Research, Sydney Children’s Hospitals Network, Westmead, New South Wales 2145, AustraliaThe University of Sydney, Faculty of Medicine and Health, Specialty of Child and Adolescent Health, Westmead, New South Wales 2145, Australia; The Australian Paediatric Surveillance Unit, Kids Research, Sydney Children’s Hospitals Network, Westmead, New South Wales 2145, Australia; The Sydney Children’s Hospitals Network, Westmead, New South Wales 2145, AustraliaIntroduction: Children living with rare diseases (RD) require considerable support from their families. Aim: To assess the impact of children’s RD on family functioning and evaluate predictors of the level of impact. Methods: Families of Australian children aged < 19 years and living with a RD were invited to complete a comprehensive survey. Impact on family (IOF) score, using the validated IOF scale, embedded within the survey, was used as the main outcome measure. Results: Families of 462 children with > 240 different RD completed the survey. IOF scores increased as child health functioning (HF) worsened (Odds ratio [OR]= 97.3 [95 % CI 40.6–233.4] for severely reduced compared to excellent HF). Speaking a first language other than English (OR 2.9; 95 % CI 1.4–6.3), experiencing financial difficulty (OR 2.9; 95 % CI 1.4–3.1), long wait times to see a specialist doctor (OR 1.9; 95 % CI 1.3–2.9), and having two versus one child with a RD (OR 3.8; 95 % CI 1.1–14.5) were also associated with higher IOF scores. Conversely, IOF scores significantly decreased as the age of children increased (6–11 y: OR 0.6 (95 % CI 0.4–0.9); 12–18 y: OR 0.5 (95 % CI 0.3–0.8), versus 0–5 y). Conclusion: Worse health-related functioning in children with RD results in significantly higher impacts on family functioning. Practical, financial, and socio-emotional support is required to reduce the burden for families whose children live with a RD, particularly families from non-English speaking backgrounds, with financial stress, or with young children who have severe disease.http://www.sciencedirect.com/science/article/pii/S2950008725000109Caregiver burdenChildFamilyRare diseases |
| spellingShingle | Judith M. Jobling Suzy M. Teutsch Guy D. Eslick Elizabeth J. Elliott Impact on the family of a child’s rare disease: A large cross-sectional study Rare Caregiver burden Child Family Rare diseases |
| title | Impact on the family of a child’s rare disease: A large cross-sectional study |
| title_full | Impact on the family of a child’s rare disease: A large cross-sectional study |
| title_fullStr | Impact on the family of a child’s rare disease: A large cross-sectional study |
| title_full_unstemmed | Impact on the family of a child’s rare disease: A large cross-sectional study |
| title_short | Impact on the family of a child’s rare disease: A large cross-sectional study |
| title_sort | impact on the family of a child s rare disease a large cross sectional study |
| topic | Caregiver burden Child Family Rare diseases |
| url | http://www.sciencedirect.com/science/article/pii/S2950008725000109 |
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