title: Zoledronic acid treatment in children with osteogenesis imperfecta.
abstract: BACKGROUND
  Intravenous disodium pamidronate has become an established treatment in osteogenesis imperfecta (OI). Another bisphosphonate, zoledronic acid, has been indicated for the treatment of adult osteoporosis. We studied its efficacy and safety in children with mild OI.
  
  
  METHODS
  Patients were treated for 1.0-3.2 years with 0.05 mg/kg zoledronic acid intravenously every 6 months as part of their clinical care. They were carefully followed for clinical and biochemical parameters, side effects, bone mineral densities (BMD) and compression fractures.
  
  
  RESULTS
  The study included 17 patients (age 1.5-16.8 years) with type I OI. They had sustained altogether 73 fractures; 9 had compression fractures. During the treatment, 6 patients suffered in total 10 new long-bone fractures. The median lumbar spine areal BMD z-score increased from -2.0 to -0.7 during 2 years of treatment. The infusions were associated with a transient decrease in serum calcium and phosphate and a significant increase in serum PTH. Two patients developed symptomatic hypocalcemia. Bone turnover markers decreased during the treatment.
  
  
  CONCLUSIONS
  Intravenous zoledronic acid is an effective mode of treatment in children with OI. The treatment response is comparable to pamidronate but the infusion protocol is more convenient. Further studies are needed to establish optimal dosing and long-term safety.
authors.affiliation: Hospital for Children and Adolescents, Pediatric Endocrinology and Metabolic Bone Diseases, University of Helsinki, Helsinki, Finland.
authors.forename: Ilkka
authors.initials: I
authors.lastname: Vuorimies
authors.forename: Sanna
authors.initials: S
authors.lastname: Toiviainen-Salo
authors.forename: Matti
authors.initials: M
authors.lastname: Hero
authors.forename: Outi
authors.initials: O
authors.lastname: Mäkitie
mesh_terms: D000293:Adolescent; D015519:Bone Density / Q000187:drug effects; D050071:Bone Density Conservation Agents / Q000627:therapeutic use; D002648:Child; D002675:Child, Preschool; D004164:Diphosphonates / Q000627:therapeutic use*; D005260:Female; D005500:Follow-Up Studies; D050723:Fractures, Bone / Q000453:epidemiology / Q000517:prevention & control; D006801:Humans; D007093:Imidazoles / Q000627:therapeutic use*; D007223:Infant; D008159:Lumbar Vertebrae / Q000187:drug effects; D008297:Male; D010013:Osteogenesis Imperfecta / Q000188:drug therapy*; D016103:Spinal Fractures / Q000453:epidemiology / Q000517:prevention & control; D016896:Treatment Outcome; D000077211:Zoledronic Acid
chemical_list: D050071:Bone Density Conservation Agents; D004164:Diphosphonates; D007093:Imidazoles; D000077211:Zoledronic Acid