Atypical femur fractures are rare, low-energy fractures that involve a specific constellation of radiographic findings. These fractures have been well described in adult osteoporotic patients on long-term bisphosphonates; however, little to no literature exists on atypical femur fractures in pediatric patients on long-term bisphosphonates. The use of bisphosphonates as treatment of osteogenesis imperfecta is common to reduce fracture rate and improve bone mineral density. We describe a 15-year-old adolescent boy with type I osteogenesis imperfecta on long-term bisphosphonate therapy. He presented with an atypical right femur fracture and an impending left femur fracture. To the authors’ knowledge, these findings represent the first case of an atypical femur fracture with a contralateral impending atypical femur fracture in a pediatric patient on long-term bisphosphonate treatment. This case highlights the importance of evaluating pediatric patients for bisphosphonate-associated complications, as is typical in adult patients. Physicians should carefully weigh the risks and benefits of bisphosphonate therapy in pediatric patients to better understand the potential adverse effects.

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