A case of a severe reaction following the use of bisphosphonates in a patient with osteogenesis imperfecta

摘要

We present a case of an unusual delayed multi-systemic reaction, following treatment with pamidronate. Although serious adverse reactions have been reported with pamidronate use, such a severe reaction, late in the course of pamidronate treatment, has not been described before. An 11-month-old boy with severe and complex osteogenesis imperfecta (OI) presented with hyperpyrexia and respiratory distress 10 days after his fifth cycle of pamidronate. He had significant derangement of his biochemical parameters including a positive urine myoglobin. His respiratory distress was out of proportion to his chest radiograph changes. Bilevel positive airway pressure (BiPAP) and paediatric intensive care (PICU) admission were required. He was extensively investigated to exclude other diagnoses, but all of these investigations were negative. The reaction resembled rhabdomyolysis. He made a full recovery with only supportive management.