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

Indian Pediatr. 2016 Mar;53(3):250-2. doi: 10.1007/s13312-016-0830-3.

Abstract

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.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Bony Callus / diagnostic imaging
  • Bony Callus / drug effects*
  • Bony Callus / pathology*
  • Diphosphonates / adverse effects*
  • Diphosphonates / therapeutic use
  • Femur / drug effects
  • Femur / pathology
  • Fractures, Bone / diagnostic imaging
  • Fractures, Bone / pathology
  • Humans
  • Hyperplasia
  • Male
  • Osteogenesis Imperfecta / drug therapy*

Substances

  • Diphosphonates

Supplementary concepts

  • Osteogenesis Imperfecta, Type V