LAUSR

PURPOSE To determine whether there was a reduction in fracture incidence amongst children with OI who were treated with both bisphosphonates and orthoses. OBJECTIVE Was there an additional reduction in fracture incidence amongst children with Osteogenesis Imperfecta (OI) treated with both bisphosphonates and Hip-Knee-Ankle-Foot-Orthosis (HKAFO)? MATERIALS AND METHODS Of the 129 OI patients treated from 1990 to 2017, retrospective data from 48 patients who participated in the bisphosphonates-orthosis regime were analyzed including the incidence of fractures and modalities of fracture treatment. RESULTS Bisphosphonates usage was more frequent than bracing and there were more positive changes (smaller or equal number of fractures each year) than negative changes (more fractures each year); negative changes were scarce, explained by non-compliance with the use of bracing. Poisson regression models were significant for positive changes, whereas the interaction between them was borderline significant. The main finding is that the association between bisphosphonates usage and the number of positive changes was stronger among the patients who used braces more frequently and weaker among patients who used bracing less frequently. CONCLUSIONS Bracing of OI patients has an additive effect on bisphosphonate treatment in fracture prevention which should lead to the reconsideration of a hybrid approach to OI management.Implications for rehabilitationTwo key goals of treatment of Osteogenesis Imperfecta (OI) patients include decreasing fracture incidence and improving function and independence as supported by a multi-disciplinary approach that combines medical, orthopaedic and rehabilitation treatments.Although the literature provides evidence that bisphosphonates reduce the frequency of fractures, there have not been reports of its effect when used with orthoses.Orthoses for OI patients have an additive effect on bisphosphonate treatment in fracture prevention.These results contribute to making an informed decision regarding this hybrid approach to OI management.