title: EFFECTS OF LONG-TERM ALENDRONATE TREATMENT ON A LARGE SAMPLE OF PEDIATRIC PATIENTS WITH OSTEOGENESIS IMPERFECTA.
abstract: Osteogenesis imperfecta (OI) is a group of inherited diseases characterized by reduced bone mass, recurrent bone fractures, and progressive bone deformities. Here, we evaluate the efficacy and safety of long-term treatment with alendronate in a large sample of Chinese children and adolescents with OI.
  
  
  
  In this prospective study, a total of 91 children and adolescents with OI were included. The patients received 3 years' treatment with 70 mg alendronate weekly and 500 mg calcium daily. During the treatment, fracture incidence, bone mineral density (BMD), and serum levels of the bone turnover biomarkers (alkaline phosphatase [ALP] and cross-linked C-telopeptide of type I collagen [β-CTX]) were evaluated. Linear growth speed and parameters of safety were also measured.
  
  
  
  After 3 years of treatment, the mean annual fracture incidence decreased from 1.2 ± 0.8 to 0.2 ± 0.3 (P<.01). BMD at the lumbar spine and femoral neck significantly increased by 74.6% and 39.5%, with their BMD Z-score increasing from -3.0 to 0.1 and from -4.2 to -1.3, respectively (both P<.01 vs. baseline). In addition, serum ALP and β-CTX levels decreased by 35.6% and 44.3%, respectively (both P<.05 vs. baseline). Height significantly increased, but without an obvious increase in its Z-score. Patient tolerance of alendronate was good.
  
  
  
  Three years' treatment with alendronate was demonstrated for the first time to significantly reduce fracture incidence, increase lumbar spine and femoral neck BMD, and decrease bone turnover biomarkers in Chinese children and adolescents with OI.
  
  
  
  ALP = alkaline phosphatase β-CTX = cross-linked C-telopeptide of type I collagen BMD = bone mineral density BP = bisphosphonate DXA = dual-energy X-ray absorptiometry 25OHD = 25-hydroxyvitamin D OI = osteogenesis imperfecta PTH = parathyroid hormone.
authors.forename: Fang
authors.initials: F
authors.lastname: Lv
authors.forename: Yi
authors.initials: Y
authors.lastname: Liu
authors.forename: Xiaojie
authors.initials: X
authors.lastname: Xu
authors.forename: Jianyi
authors.initials: J
authors.lastname: Wang
authors.forename: Doudou
authors.initials: D
authors.lastname: Ma
authors.forename: Yan
authors.initials: Y
authors.lastname: Jiang
authors.forename: Ou
authors.initials: O
authors.lastname: Wang
authors.forename: Weibo
authors.initials: W
authors.lastname: Xia
authors.forename: Xiaoping
authors.initials: X
authors.lastname: Xing
authors.forename: Wei
authors.initials: W
authors.lastname: Yu
authors.forename: Mei
authors.initials: M
authors.lastname: Li
mesh_terms: D000293:Adolescent; D019386:Alendronate / Q000008:administration & dosage / Q000494:pharmacology*; D015415:Biomarkers / Q000097:blood; D015519:Bone Density / Q000187:drug effects*; D050071:Bone Density Conservation Agents / Q000008:administration & dosage / Q000494:pharmacology*; D016723:Bone Remodeling / Q000187:drug effects*; D002648:Child; D005260:Female; D005500:Follow-Up Studies; D050723:Fractures, Bone / Q000517:prevention & control*; D006801:Humans; D008297:Male; D010013:Osteogenesis Imperfecta / Q000097:blood / Q000000981:diagnostic imaging / Q000188:drug therapy*; D017063:Outcome Assessment, Health Care; D013997:Time Factors
chemical_list: D015415:Biomarkers; D050071:Bone Density Conservation Agents; D019386:Alendronate