title: Two years of growth hormone treatment in adults with Prader-Willi syndrome do not improve the low BMD.
abstract: BACKGROUND
  Bone mineral density (BMD) in adult patients with Prader-Willi syndrome (PWS) might be low due to high bone turnover.
  
  
  OBJECTIVE
  The objective of the study was to investigate bone mass in a group of adult PWS subjects and study the effects of GH treatment on BMD and markers of bone turnover.
  
  
  METHODS
  Forty-six adults with genetically verified PWS were randomized to GH or placebo for 12 months, followed by open prospective GH for 24 additional months. BMD at the lumbar spine (LS) L1-4, the total hip, and the total body was assessed by dual-energy x-ray absorptiometry at baseline and every 12th month thereafter. Markers of bone turnover were measured at baseline and at the end of the controlled study.
  
  
  RESULTS
  In this cohort of adult subjects with PWS, baseline BMD was reduced in all compartments compared with the reference (Z-scores). Men had lower Z-scores BMD than women in LS and total body (P < .05). With 12 months of GH, LS-BMD was significantly reduced compared with placebo. No changes in BMD were observed with continuous GH treatment for 24 months. The bone formation markers increased with GH therapy compared with placebo, whereas the resorption marker did not change.
  
  
  CONCLUSIONS
  Adult PWS subjects, especially the men, have low bone mass that was not improved with GH treatment for 2 years. Because PWS subjects are short, BMD might be underestimated and should be adjusted for. Further studies, with adequate GH and sex hormone replacement throughout puberty and early adult life, are needed to better characterize PWS.
authors: Section for Specialized Endocrinology, Department of Endocrinology, Oslo University Hospital, Rikshospitalet, Oslo, Norway. andjoe@ous-hf.no
authors: A P
authors: AP
authors: Jørgensen
authors: T
authors: T
authors: Ueland
authors: R
authors: R
authors: Sode-Carlsen
authors: T
authors: T
authors: Schreiner
authors: K F
authors: KF
authors: Rabben
authors: S
authors: S
authors: Farholt
authors: C
authors: C
authors: Høybye
authors: J S
authors: JS
authors: Christiansen
authors: J
authors: J
authors: Bollerslev
mesh_terms: D015502:Absorptiometry, Photon; D000328:Adult; D015519:Bone Density / Q000187:drug effects*; D015331:Cohort Studies; D003718:Denmark; D005260:Female; D019382:Human Growth Hormone / Q000494:pharmacology / Q000627:therapeutic use*; D006801:Humans; D008159:Lumbar Vertebrae / Q000187:drug effects; D008297:Male; D009664:Norway; D010919:Placebos; D011218:Prader-Willi Syndrome / Q000188:drug therapy* / Q000378:metabolism; D013997:Time Factors; D055815:Young Adult
chemical_list: D010919:Placebos; D019382:Human Growth Hormone