title: Efficacy and Safety of Bisphosphonate Therapy in Children with Osteogenesis Imperfecta: A Systematic Review.
abstract: OBJECTIVE
  To systematically assess contemporary knowledge regarding the effectiveness and safety of bisphosphonates (BPs) in children with osteogenesis imperfecta (OI).
  
  
  METHODS
  PubMed/MEDLINE, Embase, and Cochrane were searched for eligible articles up to June 2014. Studies eligible for inclusion were (randomized) controlled trials assessing the effects of BPs in children with OI. Methodological quality was assessed independently by 4 reviewers using the Cochrane Collaboration's tool for risk of bias.
  
  
  RESULTS
  Ten studies (519 children) were included. Four studies (40%) showed a low risk of bias. All studies investigating lumbar spine areal bone mineral density indicated a significant increase as a result of BP treatment. Most studies observed a significant decrease in fracture incidence. The most frequently reported adverse events were gastrointestinal complaints, fever, and muscle soreness. A significant decrease in (bone) pain due to BP treatment was observed in more than half of the studies. Most studies measuring urinary markers of bone resorption reported a significant decrease. The majority of studies with intravenous treatment showed a significant increase in lumbar projection area, whereas studies with oral treatment did not.
  
  
  CONCLUSIONS
  Treatment with oral or intravenous BPs in children with OI results in an increase in bone mineral density and seems to be safe and well tolerated.
authors.affiliation: Child Development & Exercise Center, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht, The Netherlands.
authors.forename: Ester B G
authors.initials: EB
authors.lastname: Rijks
authors.forename: Bart C
authors.initials: BC
authors.lastname: Bongers
authors.forename: Marloes J G
authors.initials: MJ
authors.lastname: Vlemmix
authors.forename: Annemieke M
authors.initials: AM
authors.lastname: Boot
authors.forename: Atty T H
authors.initials: AT
authors.lastname: van Dijk
authors.forename: Ralph J B
authors.initials: RJ
authors.lastname: Sakkers
authors.forename: Marco
authors.initials: M
authors.lastname: van Brussel
mesh_terms: D000293:Adolescent; D002648:Child; D002675:Child, Preschool; D004164:Diphosphonates / Q000009:adverse effects* / Q000627:therapeutic use*; D005260:Female; D006801:Humans; D008297:Male; D010013:Osteogenesis Imperfecta / Q000188:drug therapy* / Q000378:metabolism / Q000473:pathology
chemical_list: D004164:Diphosphonates