title: Early bisphosphonate treatment in infants with severe osteogenesis imperfecta.
abstract: OBJECTIVE
  To evaluate prospectively the efficacy of bisphosphonate treatment in infants with severe forms of osteogenesis imperfecta (OI).
  
  
  METHODS
  Of 10 children (6 females) with OI type III, 5 (group A) started treatment (2 mg/kg neridronate administered intravenously for 2 consecutive days, every 3 months) just after diagnosis at birth and 5 (group B) after 6 months. Ten untreated children, matched for sex, age, and clinical severity of OI, constituted a historical control group (group C). We measured weight, length, and number of fractures every 3 months and serum and urinary levels of calcium, phosphorus, creatinine, serum alkaline phosphatase, 25-hydroxyvitamin D, insulin-like growth factor I, parathyroid hormone, and osteocalcin, urinary type I collagen N-terminal telopeptide, and lateral radiography of vertebral column every 6 months.
  
  
  RESULTS
  Group A had better growth and a lower incidence of fractures than groups B and C in the first 6 months of treatment. In the second 6 months, both groups A and B had lower fracture rates than group C. After 12 months of therapy, osteocalcin and insulin-like growth factor I levels significantly increased only in group A. The urinary Ca/Cr ratio and N-terminal telopeptide/Cr ratio significantly declined only in treated patients. Vertebral body area and the structure of vertebral bodies improved in all treated patients, but especially in group A.
  
  
  CONCLUSIONS
  Cyclical neridronate treatment, started just after diagnosis at birth, had positive effects on growth and fracture rate.
authors.affiliation: Pediatric Clinic and Rheumatological Rehabilitation, University of Verona, Verona, Italy. franco.antoniazzi@univr.it
authors.forename: Franco
authors.initials: F
authors.lastname: Antoniazzi
authors.forename: Giorgio
authors.initials: G
authors.lastname: Zamboni
authors.forename: Silvana
authors.initials: S
authors.lastname: Lauriola
authors.forename: Luisa
authors.initials: L
authors.lastname: Donadi
authors.forename: Silvano
authors.initials: S
authors.lastname: Adami
authors.forename: Luciano
authors.initials: L
authors.lastname: Tatò
mesh_terms: D015502:Absorptiometry, Photon; D000886:Anthropometry; D002118:Calcium / Q000097:blood / Q000652:urine; D004164:Diphosphonates / Q000627:therapeutic use*; D004334:Drug Administration Schedule; D042241:Early Diagnosis; D005260:Female; D005500:Follow-Up Studies; D050723:Fractures, Bone / Q000453:epidemiology; D006801:Humans; D015994:Incidence; D007223:Infant; D007231:Infant, Newborn; D008159:Lumbar Vertebrae / Q000000981:diagnostic imaging; D008297:Male; D015675:Osteocalcin / Q000097:blood / Q000652:urine; D010013:Osteogenesis Imperfecta / Q000175:diagnosis* / Q000188:drug therapy* / Q000453:epidemiology; D010710:Phosphates / Q000097:blood / Q000652:urine; D015995:Prevalence; D011446:Prospective Studies; D012720:Severity of Illness Index; D013997:Time Factors; D016896:Treatment Outcome
chemical_list: D004164:Diphosphonates; D010710:Phosphates; D015675:Osteocalcin; C053389:6-amino-1-hydroxyhexane-1,1-diphosphonate; D002118:Calcium