March 08, 2017
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Prior bisphosphonate use unrelated to atypical femur fractures in osteogenesis imperfecta
Among children with osteogenesis imperfecta, the occurrence of atypical femur fractures is not associated with bisphosphonate treatment history, according to findings published in the Journal of Bone and Mineral Research.
Frank Rauch, MD, of the Shriners Hospital for Children in Montreal, and colleagues evaluated 166 femur fractures among 119 children with osteogenesis imperfecta to determine whether femur fractures are more frequent in patients who received bisphosphonate therapy.
Overall, 130 fractures among 90 patients occurred in femurs with major anatomical abnormalities; in nondeformed femurs, 36 fractures occurred among 30 participants (mean age at fracture, 2.7 years). Most fractures occurred in participants with osteogenesis imperfecta type one (n = 14), followed by type four (n = 9), type six (n = 5) and types five and seven (one in each). Overall, 25 fractures occurred in the absence of prior bisphosphonate treatment and eight resembled atypical femur fractures; 11 occurred during the use of bisphosphonate treatment and three resembled atypical femur fractures.
“We observed an atypical appearance in about a quarter of nondeformed femur fractures that occurred in children with [osteogenesis imperfecta],” the researchers wrote. “Such atypical femur fractures seemed to be related to the severity of [osteogenesis imperfecta] rather than to bisphosphonate treatment history. In fractures that occurred in patients with femur deformities a transverse fracture pattern was also associated with disease severity. For further clarification, larger and prospective studies on fracture pattern and fracture epidemiology in [osteogenesis imperfecta] seem warranted.” – by Amber Cox
Disclosure: Rauch reports various financial ties with Alexion, Genzyme and Novartis. Please see the full study for a list of all other authors’ relevant financial disclosures.
Perspective
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PERSPECTIVE
Outi Mäkitie
There have been concerns regarding the long-term safety of bisphosphonates in children with osteogenesis imperfecta. In adults treated for osteoporosis, bisphosphonates have in some cases caused atypical femoral fractures. Trejo and colleagues have evaluated how bisphosphonate treatment influences characteristics of diaphyseal femur fractures in children with osteogenesis imperfecta. Of the 36 fractures in nondeformed femurs in 30 children with the disorder (age at fracture between 1 month and 17.4 years), 11 fractures in nine children occurred during bisphosphonate treatment. Three of the fractures (27%) resembled atypical femur fractures. However, similar fractures were equally common in patients without prior bisphosphonate treatment. The authors conclude that such “atypical femur fractures” seem to be related to the severity of the underlying disease rather than bisphosphonate treatment.
A similar study evaluated 127 femoral fractures in 24 children with osteogenesis imperfecta (Vuorimies I, et al. J Clin Endocrinol Metab. 2017;doi:10.1210/jc.2016-3745). The findings indicated that the characteristics or location of fractures were not influenced by bisphosphonate treatment, and there was no evidence of atypical femoral fractures in bisphosphonate-treated children with osteogenesis imperfecta. In contrast, severity of osteogenesis imperfecta correlated with fracture characteristics; distal location and transverse configuration were more common in the more severe types compared with the mildest form, type 1.
Based on these two independent studies, bisphosphonate treatment in children with osteogenesis imperfecta does not cause atypical femoral fractures.
Outi Mäkitie, MD, PhD
Professor of Pediatric Endocrinology, Children’s Hospital, University of Helsinki and Helsinki University Hospital, Helsinki, Finland
Disclosure: Mäkitie reports receiving honoraria, fees for consulting and lectures, and travel support from Alexion and Kyowa Kirin.
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