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Recurrent Proximal Femur Fractures in a Teenager with Osteogenesis Imperfecta on Continuous Bisphosphonate Therapy: Are we Overtreating?

著者 Vasanwala RF , Sanghrajka A , Bishop NJ , Högler W
J Bone Miner Res.2016 Feb 4 ; ():.
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Long-term bisphosphonate therapy in adults with osteoporosis is associated with atypical femoral fractures, caused by increased material bone density and prolonged suppression of bone remodeling which may reduce fracture toughness. In children with osteogenesis imperfecta, long-term intravenous bisphosphonate therapy improves bone structure and mass without further increasing the already hypermineralised bone matrix, and is generally regarded as safe. Here we report a teenage girl with osteogenesis imperfecta type IV, who was started on cyclical intravenous pamidronate therapy at age 6 years because of recurrent fractures. Trans-iliac bone biopsy revealed classical structural features of OI but unusually low bone resorption surfaces. She made substantial improvements in functional ability, bone mass and fracture rate. However, after 5 years of pamidronate therapy she started to develop recurrent, bilateral, non-traumatic, proximal femur fractures which satisfied the case definition for atypical femur fractures. Some fractures were preceded by periosteal reactions and prodromal pain. Pamidronate was discontinued after 7 years of therapy, following which she sustained two further non-traumatic femur fractures, and continued to show delayed tibial osteotomy healing. Despite rodding surgery, and very much in contrast to her affected, untreated and normally mobile mother, she remains wheelchair dependent. The case of this girl raises questions about the long-term safety of bisphosphonate therapy in some children, in particular about the risk of oversuppressed bone remodeling with the potential for micro-crack accumulation, delayed healing and increased stiffness. The principal concern is whether there is point at which benefit from bisphosphonate therapy could turn into harm, where fracture risk increases again. This case should stimulate debate whether current adult atypical femoral fracture guidance should apply to children, and whether low-frequency, low-dose cyclical, intermittent or oral treatment maintenance regimens should be considered on a case-by-case basis. This article is protected by copyright. All rights reserved.
PMID: 26845496 [PubMed - as supplied by publisher]
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