Medication-related osteonecrosis of the jaw in an adult with osteogenesis imperfecta: a case report

Bisphosphonates (BPs) are widely used for managing tumor metastasis and bone diseases, associated with a rare complication of medication-related osteonecrosis of the jaws (MRONJ). Though reasons are still debated, the MRONJ occurrence in paediatric population suffering osteogenesis imperfecta (OI) and treated with BPs is absent. Here, we reported a 33-year-old patient with OI (Type III) who developed MRONJ after receiving intravenous pamidronate from 16 years old. She complained of intermittent nasal bleeding for six years, and presented an exposed necrotic bone in the left maxilla. She underwent surgical debridement, and pathological examination confirmed MRONJ. To our knowledge, this was a very rare case of MRONJ in an adult with OI type III following pediatric BP exposure. This case underscored the need for long-term vigilance, as delayed onset into adulthood is possible. Clinicians should pay attention to sinonasal symptoms as potential manifestations of MRONJ, and strengthen dental preventive care to reduce local risk factors. Long-term follow-up longitudinal studies with a larger sample size in this population are needed.