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 |
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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 – sequence: 3 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 – sequence: 5 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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| CitedBy_id | crossref_primary_10_1111_cen_14372 crossref_primary_10_1016_j_bonr_2023_101700 crossref_primary_10_1210_clinem_dgac556 crossref_primary_10_3389_fendo_2024_1382583 crossref_primary_10_3390_jcm10194361 crossref_primary_10_1186_s13023_020_01651_x crossref_primary_10_1210_clinem_dgab406 crossref_primary_10_1016_j_npep_2020_102084 crossref_primary_10_1159_000518342 crossref_primary_10_1080_17446651_2021_1898375 crossref_primary_10_3390_endocrines3020027 crossref_primary_10_1097_MED_0000000000000517 crossref_primary_10_3390_ijms251810169 crossref_primary_10_1186_s13633_020_00090_6 crossref_primary_10_3390_jpm12060858 crossref_primary_10_3390_ijms22052705 |
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| Keywords | adult height sex steroid replacement therapy bone mineral density growth hormone treatment Prader-Willi syndrome young adults |
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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 |
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