Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial
Abstract Background Osteogenesis Imperfecta (OI) is a rare genetic disorder characterized by bone fragility and susceptibility to fractures. No curative treatment currently exists, and limited data are available on the effects of adapted physical activity (APA). This study evaluates the impact of AP...
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BMC
2025-04-01
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| Series: | Orphanet Journal of Rare Diseases |
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| Online Access: | https://doi.org/10.1186/s13023-025-03678-4 |
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| author | Hayssam Al Arab Sacha Flammier Morgane Espitalier Justine Bacchetta Marine Fouillet-Desjonqueres |
| author_facet | Hayssam Al Arab Sacha Flammier Morgane Espitalier Justine Bacchetta Marine Fouillet-Desjonqueres |
| author_sort | Hayssam Al Arab |
| collection | DOAJ |
| description | Abstract Background Osteogenesis Imperfecta (OI) is a rare genetic disorder characterized by bone fragility and susceptibility to fractures. No curative treatment currently exists, and limited data are available on the effects of adapted physical activity (APA). This study evaluates the impact of APA on bone health, physical function, respiratory function, and quality of life in pediatric children with OI. Methods The MOVE-OI trial (NCT 04119388) is a prospective single-center study assessing the impact of a 12-month individualized APA program. Inclusion criteria included confirmed OI pathogenic variant, ages 6–18 years. Baseline (M0) and end-point (M12) assessments included clinical, radiological, and respiratory evaluations. The primary outcome was an improvement in the 6-min walk test (6MWT) distance. A non-parametric paired-test was performed for analysis. Results Thirty participants (16 males, median age 10.5 years) completed the program. A 17% increase in the 6MWT distance (p = 0.0007) was observed, with an average improvement of 98 m. No significant bone density or respiratory function changes were detected. Fracture incidence decreased (from 40 to 20%), and quality-of-life improvements were noted in participants with high baseline difficulty scores. Conclusion APA improves endurance and physical capacity in children with OI. Multidisciplinary care and further research are needed to enhance long-term outcomes. |
| format | Article |
| id | doaj-art-608a2bd9ce514993abbfb5c5f1f0b974 |
| institution | OA Journals |
| issn | 1750-1172 |
| language | English |
| publishDate | 2025-04-01 |
| publisher | BMC |
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| series | Orphanet Journal of Rare Diseases |
| spelling | doaj-art-608a2bd9ce514993abbfb5c5f1f0b9742025-08-20T02:12:02ZengBMCOrphanet Journal of Rare Diseases1750-11722025-04-012011910.1186/s13023-025-03678-4Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trialHayssam Al Arab0Sacha Flammier1Morgane Espitalier2Justine Bacchetta3Marine Fouillet-Desjonqueres4Department of Pediatric Nephrology, Rheumatology and Dermatology, Hôpital Femme Mère EnfantDepartment of Pediatric Nephrology, Rheumatology and Dermatology, Hôpital Femme Mère EnfantDepartment of Pediatric Nephrology, Rheumatology and Dermatology, Hôpital Femme Mère EnfantDepartment of Pediatric Nephrology, Rheumatology and Dermatology, Hôpital Femme Mère EnfantDepartment of Pediatric Nephrology, Rheumatology and Dermatology, Hôpital Femme Mère EnfantAbstract Background Osteogenesis Imperfecta (OI) is a rare genetic disorder characterized by bone fragility and susceptibility to fractures. No curative treatment currently exists, and limited data are available on the effects of adapted physical activity (APA). This study evaluates the impact of APA on bone health, physical function, respiratory function, and quality of life in pediatric children with OI. Methods The MOVE-OI trial (NCT 04119388) is a prospective single-center study assessing the impact of a 12-month individualized APA program. Inclusion criteria included confirmed OI pathogenic variant, ages 6–18 years. Baseline (M0) and end-point (M12) assessments included clinical, radiological, and respiratory evaluations. The primary outcome was an improvement in the 6-min walk test (6MWT) distance. A non-parametric paired-test was performed for analysis. Results Thirty participants (16 males, median age 10.5 years) completed the program. A 17% increase in the 6MWT distance (p = 0.0007) was observed, with an average improvement of 98 m. No significant bone density or respiratory function changes were detected. Fracture incidence decreased (from 40 to 20%), and quality-of-life improvements were noted in participants with high baseline difficulty scores. Conclusion APA improves endurance and physical capacity in children with OI. Multidisciplinary care and further research are needed to enhance long-term outcomes.https://doi.org/10.1186/s13023-025-03678-4Osteogenesis imperfecta (OI)Bone fragilityAdapted physical activity (APA)Pediatric children6-min walk testProspective trial |
| spellingShingle | Hayssam Al Arab Sacha Flammier Morgane Espitalier Justine Bacchetta Marine Fouillet-Desjonqueres Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial Orphanet Journal of Rare Diseases Osteogenesis imperfecta (OI) Bone fragility Adapted physical activity (APA) Pediatric children 6-min walk test Prospective trial |
| title | Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial |
| title_full | Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial |
| title_fullStr | Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial |
| title_full_unstemmed | Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial |
| title_short | Evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta: the MOVE-OI trial |
| title_sort | evaluation of the benefits of adapted physical activity in children and adolescents with osteogenesis imperfecta the move oi trial |
| topic | Osteogenesis imperfecta (OI) Bone fragility Adapted physical activity (APA) Pediatric children 6-min walk test Prospective trial |
| url | https://doi.org/10.1186/s13023-025-03678-4 |
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