Bone mineral density in young adults with Prader‐Willi syndrome: A randomized, placebo‐controlled, crossover GH trial

Summary Context The prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH migh...

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Veröffentlicht in:Clinical endocrinology (Oxford) Jg. 88; H. 6; S. 806 - 812
Hauptverfasser: Donze, Stephany H., Kuppens, Renske J., Bakker, Nienke E., van Alfen‐van der Velden, Janiëlle A.E.M., Hokken‐Koelega, Anita C.S.
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Sprache:Englisch
Veröffentlicht: England Wiley Subscription Services, Inc 01.06.2018
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ISSN:0300-0664, 1365-2265, 1365-2265
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Abstract Summary Context The prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD. Objective To investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH‐treated young adults with PWS who had attained AH. Design Two‐year, randomized, double‐blind, placebo‐controlled, crossover GH study. Patients Twenty‐seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m2/day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year. Measurements Bone mineral density of the total body (BMDTB) and lumbar spine (BMDLS) SDS were measured by dual‐energy x‐ray absorptiometry. Results At AH, BMDTBSDS was significantly lower compared to healthy peers (P < .01), while BMADLSSDS was similar. Both BMDTBSDS and BMADLSSDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMDTBSDS and BMADLSSDS decreased during the 2‐year study (P = .11 and P = .01), regardless of GH or placebo, while BMDTBSDS increased in those with SSRT (P < .01). Conclusions Compared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.
AbstractList Summary Context The prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD. Objective To investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH‐treated young adults with PWS who had attained AH. Design Two‐year, randomized, double‐blind, placebo‐controlled, crossover GH study. Patients Twenty‐seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m2/day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year. Measurements Bone mineral density of the total body (BMDTB) and lumbar spine (BMDLS) SDS were measured by dual‐energy x‐ray absorptiometry. Results At AH, BMDTBSDS was significantly lower compared to healthy peers (P < .01), while BMADLSSDS was similar. Both BMDTBSDS and BMADLSSDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMDTBSDS and BMADLSSDS decreased during the 2‐year study (P = .11 and P = .01), regardless of GH or placebo, while BMDTBSDS increased in those with SSRT (P < .01). Conclusions Compared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.
The prevalence of osteoporosis is increased in adults with Prader-Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD. To investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH-treated young adults with PWS who had attained AH. Two-year, randomized, double-blind, placebo-controlled, crossover GH study. Twenty-seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m /day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year. Bone mineral density of the total body (BMD ) and lumbar spine (BMD ) SDS were measured by dual-energy x-ray absorptiometry. At AH, BMD SDS was significantly lower compared to healthy peers (P < .01), while BMAD SDS was similar. Both BMD SDS and BMAD SDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMD SDS and BMAD SDS decreased during the 2-year study (P = .11 and P = .01), regardless of GH or placebo, while BMD SDS increased in those with SSRT (P < .01). Compared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.
The prevalence of osteoporosis is increased in adults with Prader-Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD.CONTEXTThe prevalence of osteoporosis is increased in adults with Prader-Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD.To investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH-treated young adults with PWS who had attained AH.OBJECTIVETo investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH-treated young adults with PWS who had attained AH.Two-year, randomized, double-blind, placebo-controlled, crossover GH study.DESIGNTwo-year, randomized, double-blind, placebo-controlled, crossover GH study.Twenty-seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m2 /day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year.PATIENTSTwenty-seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m2 /day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year.Bone mineral density of the total body (BMDTB ) and lumbar spine (BMDLS ) SDS were measured by dual-energy x-ray absorptiometry.MEASUREMENTSBone mineral density of the total body (BMDTB ) and lumbar spine (BMDLS ) SDS were measured by dual-energy x-ray absorptiometry.At AH, BMDTB SDS was significantly lower compared to healthy peers (P < .01), while BMADLS SDS was similar. Both BMDTB SDS and BMADLS SDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMDTB SDS and BMADLS SDS decreased during the 2-year study (P = .11 and P = .01), regardless of GH or placebo, while BMDTB SDS increased in those with SSRT (P < .01).RESULTSAt AH, BMDTB SDS was significantly lower compared to healthy peers (P < .01), while BMADLS SDS was similar. Both BMDTB SDS and BMADLS SDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMDTB SDS and BMADLS SDS decreased during the 2-year study (P = .11 and P = .01), regardless of GH or placebo, while BMDTB SDS increased in those with SSRT (P < .01).Compared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.CONCLUSIONSCompared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.
ContextThe prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial effects on bone mineral density (BMD). BMD might deteriorate after cessation of GH at adult height (AH), while continuing GH might maintain BMD.ObjectiveTo investigate the effects of GH vs placebo, and furthermore the effects of sex steroid replacement therapy (SSRT), on BMD in GH‐treated young adults with PWS who had attained AH.DesignTwo‐year, randomized, double‐blind, placebo‐controlled, crossover GH study.PatientsTwenty‐seven young adults with PWS were stratified for gender and BMI and then randomly and blindly assigned to receive GH (0.67 mg/m2/day) or placebo for 1 year, after which they crossed over to the alternative treatment for another year.MeasurementsBone mineral density of the total body (BMDTB) and lumbar spine (BMDLS) SDS were measured by dual‐energy x‐ray absorptiometry.ResultsAt AH, BMDTBSDS was significantly lower compared to healthy peers (P < .01), while BMADLSSDS was similar. Both BMDTBSDS and BMADLSSDS were similar during 1 year of GH vs 1 year of placebo. In hypogonadal young adults without SSRT, BMDTBSDS and BMADLSSDS decreased during the 2‐year study (P = .11 and P = .01), regardless of GH or placebo, while BMDTBSDS increased in those with SSRT (P < .01).ConclusionsCompared to GH treatment, 1 year of placebo after attainment of AH does not deteriorate BMD SDS in young adults with PWS. In addition, our data suggest that GH is not able to prevent the decline in BMD SDS in hypogonadal young adults with PWS, unless it is combined with SSRT.
Author Donze, Stephany H.
Kuppens, Renske J.
van Alfen‐van der Velden, Janiëlle A.E.M.
Hokken‐Koelega, Anita C.S.
Bakker, Nienke E.
Author_xml – sequence: 1
  givenname: Stephany H.
  orcidid: 0000-0002-9249-4284
  surname: Donze
  fullname: Donze, Stephany H.
  email: S.Donze@kindengroei.nl
  organization: Erasmus University Medical Center‐Sophia Children's Hospital
– sequence: 2
  givenname: Renske J.
  surname: Kuppens
  fullname: Kuppens, Renske J.
  organization: Erasmus University Medical Center‐Sophia Children's Hospital
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  givenname: Nienke E.
  surname: Bakker
  fullname: Bakker, Nienke E.
  organization: Erasmus University Medical Center‐Sophia Children's Hospital
– sequence: 4
  givenname: Janiëlle A.E.M.
  surname: van Alfen‐van der Velden
  fullname: van Alfen‐van der Velden, Janiëlle A.E.M.
  organization: Radboud University Medical Center‐Amalia Children's Hospital
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  givenname: Anita C.S.
  surname: Hokken‐Koelega
  fullname: Hokken‐Koelega, Anita C.S.
  organization: Erasmus University Medical Center‐Sophia Children's Hospital
BackLink https://www.ncbi.nlm.nih.gov/pubmed/29418016$$D View this record in MEDLINE/PubMed
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2018 John Wiley & Sons Ltd.
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Keywords adult height
sex steroid replacement therapy
bone mineral density
growth hormone treatment
Prader-Willi syndrome
young adults
Language English
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Snippet Summary Context The prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has...
The prevalence of osteoporosis is increased in adults with Prader-Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has beneficial...
ContextThe prevalence of osteoporosis is increased in adults with Prader‐Willi syndrome (PWS). In children with PWS, growth hormone (GH) treatment has...
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SubjectTerms adult height
Body height
Bone density
Bone mineral density
Children
growth hormone treatment
Growth hormones
Hormone replacement therapy
Osteoporosis
Prader-Willi syndrome
sex steroid replacement therapy
Spine (lumbar)
Young adults
Title Bone mineral density in young adults with Prader‐Willi syndrome: A randomized, placebo‐controlled, crossover GH trial
URI https://onlinelibrary.wiley.com/doi/abs/10.1111%2Fcen.13567
https://www.ncbi.nlm.nih.gov/pubmed/29418016
https://www.proquest.com/docview/2047377255
https://www.proquest.com/docview/1999686088
Volume 88
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