Worsening of Callus Hyperplasia after Bisphosphonate Treatment in Type V Osteogenesis Imperfecta.

Indian Pediatr

Departments of Medical Genetics and *Orthopaedics, Nizams Institute of Medical Sciences, Hyderabad, Telangana; and Department of Medical Genetics, SGPGIMS, Lucknow, Uttar Pradesh; India. Correspondence to: Dr Prajnya Ranganath, Department of Medical Genetics, Nizams Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana 500 082, India.

Published: March 2016

Background: Type V osteogenesis imperfecta is characterized by hyperplastic callus formation and interosseus membrane calcification.

Case Characteristics: A 16-year-old boy who presented with history of recurrent fractures, had hard persistent swellings at fracture sites, and had radiographic features of hyperplastic callus and interosseus membrane calcification.

Outcome: Sequence analysis of the IFITM5 gene revealed the c.-14 C>T mutation. The patient had significant exacerbation of callus hyperplasia after initiation of bisphosphonate therapy, which reversed following cessation of the treatment.

Message: Bisphosphonates may exacerbate callus hyperplasia, and may therefore have to be used with caution in patients with type V osteogenesis imperfecta.

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http://dx.doi.org/10.1007/s13312-016-0830-3DOI Listing

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