Fatality owing to pulmonary hemorrhage following pamidronate disodium administration in a neonate with osteogenesis imperfecta type 2: A case report

• Published in: Clinical Pediatric Endocrinology Vol. 33; no. 2; pp. 76 - 81
• Main Authors: Nagoshi, Rintaro, Amari, Shoichiro, Abiko, Yu, Wada, Yuka Sano, Ishiguro, Akira, Horikawa, Reiko, Ito, Yushi
• Format: Journal Article
• Language: English
• Published: Japan The Japanese Society for Pediatric Endocrinology 2024
• Subjects: bisphosphonates; Case Report; osteogenesis imperfecta; pamidronate disodium; pulmonary hemorrhage; pulmonary hemorrhage; bisphosphonates; osteogenesis imperfecta; pamidronate disodium
• ISSN: 0918-5739; 1347-7358
• DOI: 10.1297/cpe.2023-0061

We report the case of a patient with osteogenesis imperfecta (OI) who developed pulmonary hemorrhage 4 d after pamidronate disodium (PA) administration, despite a relatively stable respiratory status. Bisphosphonates are introduced to reduce osteoclast activity and are now widely used in patients with OI. Bisphosphonates are typically well-tolerated in children, and the standard of care involves cyclic intravenous administration of PA. However, in practice, there is limited experience with the use of PA for severe OI during the neonatal period, and its safety remains uncertain. This report aimed to describe the respiratory events potentially associated with PA in a neonatal patient with OI type 2, suggesting that serious life-threatening complications of pulmonary hemorrhage may occur after PA administration. Further studies are required to assess the relationship between pulmonary hemorrhage and PA administration, aiming to enhance prophylaxis measures.