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首页> 外文期刊>Journal of bone and mineral research: the official journal of the American Society for Bone and Mineral Research >Recurrent Proximal Femur Fractures in a Teenager With Osteogenesis Imperfecta on Continuous Bisphosphonate Therapy: Are We Overtreating?
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Recurrent Proximal Femur Fractures in a Teenager With Osteogenesis Imperfecta on Continuous Bisphosphonate Therapy: Are We Overtreating?

机译：在连续双膦酸盐疗法中复发近端股骨骨折，与骨育植物疗法：我们过度治疗吗？

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Long-term bisphosphonate (BP) 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 (OI), long-term intravenous BP therapy improves bone structure and mass without further increasing the already hypermineralized bone matrix, and is generally regarded as safe. Here we report a teenage girl with OI type IV, who was started on cyclical intravenous pamidronate therapy at age 6 years because of recurrent fractures. Transiliac 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, nontraumatic, and 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 nontraumatic 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 BP therapy in some children, in particular about the risk of oversuppressed bone remodeling with the potential for microcrack accumulation, delayed healing, and increased stiffness. The principal concern is whether there is point at which benefit from BP 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. (C) 2016 American Society for Bone and Mineral Research.
机译：具有骨质疏松症的成年人的长期双膦酸盐（BP）治疗与非典型股骨骨折有关，由增加的材料骨密度和延长抑制骨重塑引起的，这可能降低断裂韧性。在患有成骨细胞的儿童（OI）中，长期静脉注射BP疗法改善了骨骼结构和质量，而不需要进一步增加已经过高骨质化的骨基质，并且通常被认为是安全的。在这里，我们报告了一个少女患有II型的少女，由于复发性骨折，6年龄在6岁时开始于周期性静脉内氨基膦酸盐治疗。颅骨骨活检显示OI但异常低的骨吸收表面的经典结构特征。她的功能能力，骨质量和断裂率大量改善。然而，经过5年的氨化杀虫治疗，她开始开发经常性，双侧，非向量和近端股骨骨折，这使得非典型股骨骨折的情况定义。在骨膜反应和前骨疼痛之前，一些骨折。在治疗7年后停止甘草酸盐，随后她持续两种进一步的非创伤性股骨骨折，并继续显示出延迟的胫骨骨质切除术治疗。尽管罗丁手术，但与她的受影响，未经治疗的和通常移动的母亲相比，她仍然是轮椅依赖的。这个女孩的案例提出了关于一些孩子的BP疗法的长期安全的问题，特别是关于过度抑制骨重塑的风险与微裂纹积聚，延迟愈合和增加的刚度的潜力。主要关切的是，是否有植物疗法的益处可能会造成伤害，其中骨折风险再次增加。这种情况应刺激争论是否应该适用于儿童的当前成年人的非典型股骨骨折指导，以及是否应根据案例考虑低频，低剂量循环，间歇性或口服治疗维护方案。 （c）2016年美国骨骼和矿物学学会。

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