title: Bone mineral density and fracture rate in response to intravenous and oral bisphosphonates in adult osteogenesis imperfecta.
abstract: The effect of bisphosphonate treatment on bone mineral density (BMD) and fracture rates was assessed in adults with osteogenesis imperfecta (OI). This observational nonrandomized study included 90 OI adults treated with intravenous pamidronate (n = 28), oral alendronate (n = 10), or oral residronate (n = 17) or not treated (n = 35). There were 63 type I, 15 type III, and 12 type IV OI patients. BMD results were observed for up to 161 months and an average of 52 months of treatment. For type I and grouped type III/IV patients, treatment with pamidronate showed an increasing rate in L1-L4 BMD from baseline (0.006 [P = 0.03] and 0.016 [P < 0.001] gm/cm(2)/year, respectively); oral bisphosphonate treatment showed a significant increasing rate in L1-L4 BMD (0.004 gm/cm(2)/year [P = 0.047]) for type I patients. Pamidronate-treated type III/IV and oral bisphosphonate-treated type I patients showed significant increases in total-hip BMD (0.006 [P = 0.003] and 0.011 [P = 0.046] gm/cm(2)/year, respectively). Bisphosphonate effect on fracture rate was assessed for 5-year periods before and after treatment in 51 treated and 22 nontreated individuals matched for age at which bisphosphonate was first administered to the treated group. Bisphosphonate treatment did not decrease fracture rate in type I OI patients. Fracture rate decreased in type III/IV patients following pamidronate but not following oral bisphosphonate treatment. These results underscore a need to consider whether bisphosphonate treatment is appropriate for all adults with OI.
authors.affiliation: Osteogenesis Imperfecta Program, Kennedy Krieger Institute, 707 North Broadway, Baltimore, MD 21205, USA. shapiroj@kennedykrieger.org
authors.forename: Jay R
authors.initials: JR
authors.lastname: Shapiro
authors.forename: Carol B
authors.initials: CB
authors.lastname: Thompson
authors.forename: Yimei
authors.initials: Y
authors.lastname: Wu
authors.forename: Martin
authors.initials: M
authors.lastname: Nunes
authors.forename: Carolynn
authors.initials: C
authors.lastname: Gillen
mesh_terms: D000284:Administration, Oral; D000328:Adult; D019386:Alendronate / Q000627:therapeutic use; D015519:Bone Density / Q000187:drug effects*; D050071:Bone Density Conservation Agents / Q000008:administration & dosage* / Q000627:therapeutic use; D015331:Cohort Studies; D004164:Diphosphonates / Q000008:administration & dosage* / Q000627:therapeutic use; D005260:Female; D050723:Fractures, Bone / Q000517:prevention & control*; D006801:Humans; D007275:Injections, Intravenous; D008297:Male; D010013:Osteogenesis Imperfecta / Q000145:classification / Q000188:drug therapy*; D000077268:Pamidronate; D012189:Retrospective Studies; D016896:Treatment Outcome
chemical_list: D050071:Bone Density Conservation Agents; D004164:Diphosphonates; D000077268:Pamidronate; D019386:Alendronate