title: Fracture during intravenous bisphosphonate treatment in a child with osteogenesis imperfecta: an argument for a more frequent, low-dose treatment regimen.
abstract: OBJECTIVE
  Intravenous bisphosphonate therapy is the mainstay of medical treatment in osteogenesis imperfecta (OI) and has been shown to increase bone mass, decrease bone pain, improve mobility, and reduce the incidence of fractures. Sclerotic metaphyseal lines parallel to the growth plate are seen on long bone radiographs following cyclical intravenous therapy. These areas create stress risers within the bone that may act as foci for subsequent fractures as exemplified in this clinical case.
  
  
  METHODS
  An 8-year-old girl with OI sustained a distal radial fracture following 3 years of treatment with 6-monthly intravenous zoledronate. Her diagnosis, response to treatment, and subsequent fracture at a sclerotic metaphyseal line is described.
  
  
  RESULTS
  Peripheral quantitative computer tomography was used to characterise the presence of multiple stress risers at the distal forearm. Trabecular bone mineral density fluctuated from 34 to 126% compared to neighbouring 2-mm regions.
  
  
  CONCLUSIONS
  There remain many unanswered questions about optimal bisphosphonate treatment regimens in children with OI. The formation of stress risers following intravenous bisphosphonate treatment raises the hypothesis that a more frequent and low-dose bisphosphonate regimen would provide more uniform dosing of bone in the growing child and reduce the likelihood of fractures compared to current treatment practices.
authors.affiliation: Institute of Endocrinology and Diabetes, The Sydney Children's Hospitals Network, Westmead, N.S.W., Australia.
authors.forename: Andrew
authors.initials: A
authors.lastname: Biggin
authors.forename: Julie N
authors.initials: JN
authors.lastname: Briody
authors.forename: Elizabeth
authors.initials: E
authors.lastname: Ormshaw
authors.forename: Karen K Y
authors.initials: KK
authors.lastname: Wong
authors.forename: Bruce H
authors.initials: BH
authors.lastname: Bennetts
authors.forename: Craig F
authors.initials: CF
authors.lastname: Munns
mesh_terms: D015519:Bone Density / Q000187:drug effects*; D050071:Bone Density Conservation Agents / Q000008:administration & dosage / Q000009:adverse effects*; D002648:Child; D002675:Child, Preschool; D004164:Diphosphonates / Q000008:administration & dosage / Q000009:adverse effects*; D005260:Female; D006801:Humans; D007093:Imidazoles / Q000008:administration & dosage / Q000009:adverse effects*; D010013:Osteogenesis Imperfecta / Q000000981:diagnostic imaging / Q000188:drug therapy / Q000378:metabolism; D011885:Radius Fractures / Q000139:chemically induced / Q000000981:diagnostic imaging / Q000378:metabolism; D014057:Tomography, X-Ray Computed; D000077211:Zoledronic Acid
chemical_list: D050071:Bone Density Conservation Agents; D004164:Diphosphonates; D007093:Imidazoles; D000077211:Zoledronic Acid