Bone mineral density during 3 years of growth hormone in previously GH-treated young adults with PWS

Layla Damen; Lionne N. Grootjen; Stephany H. Donze; Laura C.G. De Graaff; Janielle A.E.M. Van Der Velden; Anita C.S. Hokken-Koelega

doi:10.1530/eje-20-1335

Bone mineral density during 3 years of growth hormone in previously GH-treated young adults with PWS

Layla Damen^*, Lionne N. Grootjen, Stephany H. Donze, Laura C.G. De Graaff, Janielle A.E.M. Van Der Velden, Anita C.S. Hokken-Koelega

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

4 Citations (Scopus)

Abstract

Objective: In children with Prader-Willi syndrome (PWS), growth hormone ( GH) treatment has positive effects on bone mineral density (BMD). Two 1-year studies did not show a differe nce between GH or placebo on BMD in young adults with PWS. However, there are no studies investigating BM D during longer-term GH treatment in young adults with PWS. Design: Open-label, a prospective study in 43 young adults with PWS. Methods: BMD of the total body (BMDTBSDS) and lumbar spine (BMADLSSDS) measured by DXA. Results: In the total group, estimated mean (95% CI) of BMD TB remained similar during 3 years of GH, being -0.76 (-1.11 to -0.41) SDS at start and -0.90 (-1.27 to -0.54) SDS after 3 years (P = 0.11), as did BMADLS, being -0.36 (-0.72 to 0.01) SDS and -0.46 (-0.77 to -0.16) SDS, respectively (P = 0.16). In men, there was a significant decrease in BMDTBSDS during 3 years of GH, while BMAD LSSDS remained similar. In women, both BMDTBSDS and BMADLSSDS remained similar. BMDTBSDS was associated with female sex, lean body mass and age. The majority of patients received sex steroid replacement therapy (SSRT). Conclusions: During 3 years of combined GH and SSRT treatment, BMD remained stable in the normal range in young adults with PWS. However, men showed a decline in BMD TBSDS, probably due to insufficient SSRT. We recommended to continue GH treatment in young adults with PWS a nd to start SSRT during adolescence unless puberty progresses normally.

Original language	English
Pages (from-to)	773-782
Number of pages	10
Journal	European Journal of Endocrinology
Volume	184
Issue number	6
DOIs	https://doi.org/10.1530/eje-20-1335
Publication status	Published - Jun 2021

Bibliographical note

Funding Information:
This study was an investigator-initiated study, supported by an independent research grant from Pfizer. Pfizer was not involved in conception or design of the study, nor in collection, analysis or interpretation of data, writing the manuscript, or decision to submit the manuscript for publication.

Publisher Copyright:
© 2021 European Society of Endocrinology.

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EMC MM-01-54-01

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This output contributes to the following UN Sustainable Development Goals (SDGs)

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10.1530/eje-20-1335

https://eje.bioscientifica.com/downloadpdf/journals/eje/184/6/EJE-20-1335.pdf

Cite this

@article{eabde6a4465c4bd9a114d42559b40b63,

title = "Bone mineral density during 3 years of growth hormone in previously GH-treated young adults with PWS",

abstract = "Objective: In children with Prader-Willi syndrome (PWS), growth hormone ( GH) treatment has positive effects on bone mineral density (BMD). Two 1-year studies did not show a differe nce between GH or placebo on BMD in young adults with PWS. However, there are no studies investigating BM D during longer-term GH treatment in young adults with PWS. Design: Open-label, a prospective study in 43 young adults with PWS. Methods: BMD of the total body (BMDTBSDS) and lumbar spine (BMADLSSDS) measured by DXA. Results: In the total group, estimated mean (95% CI) of BMD TB remained similar during 3 years of GH, being -0.76 (-1.11 to -0.41) SDS at start and -0.90 (-1.27 to -0.54) SDS after 3 years (P = 0.11), as did BMADLS, being -0.36 (-0.72 to 0.01) SDS and -0.46 (-0.77 to -0.16) SDS, respectively (P = 0.16). In men, there was a significant decrease in BMDTBSDS during 3 years of GH, while BMAD LSSDS remained similar. In women, both BMDTBSDS and BMADLSSDS remained similar. BMDTBSDS was associated with female sex, lean body mass and age. The majority of patients received sex steroid replacement therapy (SSRT). Conclusions: During 3 years of combined GH and SSRT treatment, BMD remained stable in the normal range in young adults with PWS. However, men showed a decline in BMD TBSDS, probably due to insufficient SSRT. We recommended to continue GH treatment in young adults with PWS a nd to start SSRT during adolescence unless puberty progresses normally.",

author = "Layla Damen and Grootjen, {Lionne N.} and Donze, {Stephany H.} and {De Graaff}, {Laura C.G.} and {Van Der Velden}, {Janielle A.E.M.} and Hokken-Koelega, {Anita C.S.}",

note = "Funding Information: This study was an investigator-initiated study, supported by an independent research grant from Pfizer. Pfizer was not involved in conception or design of the study, nor in collection, analysis or interpretation of data, writing the manuscript, or decision to submit the manuscript for publication. Publisher Copyright: {\textcopyright} 2021 European Society of Endocrinology.",

year = "2021",

month = jun,

doi = "10.1530/eje-20-1335",

language = "English",

volume = "184",

pages = "773--782",

journal = "European Journal of Endocrinology",

issn = "0804-4643",

publisher = "Oxford University Press",

number = "6",

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TY - JOUR

T1 - Bone mineral density during 3 years of growth hormone in previously GH-treated young adults with PWS

AU - Damen, Layla

AU - Grootjen, Lionne N.

AU - Donze, Stephany H.

AU - De Graaff, Laura C.G.

AU - Van Der Velden, Janielle A.E.M.

AU - Hokken-Koelega, Anita C.S.

N1 - Funding Information: This study was an investigator-initiated study, supported by an independent research grant from Pfizer. Pfizer was not involved in conception or design of the study, nor in collection, analysis or interpretation of data, writing the manuscript, or decision to submit the manuscript for publication. Publisher Copyright: © 2021 European Society of Endocrinology.

PY - 2021/6

Y1 - 2021/6

N2 - Objective: In children with Prader-Willi syndrome (PWS), growth hormone ( GH) treatment has positive effects on bone mineral density (BMD). Two 1-year studies did not show a differe nce between GH or placebo on BMD in young adults with PWS. However, there are no studies investigating BM D during longer-term GH treatment in young adults with PWS. Design: Open-label, a prospective study in 43 young adults with PWS. Methods: BMD of the total body (BMDTBSDS) and lumbar spine (BMADLSSDS) measured by DXA. Results: In the total group, estimated mean (95% CI) of BMD TB remained similar during 3 years of GH, being -0.76 (-1.11 to -0.41) SDS at start and -0.90 (-1.27 to -0.54) SDS after 3 years (P = 0.11), as did BMADLS, being -0.36 (-0.72 to 0.01) SDS and -0.46 (-0.77 to -0.16) SDS, respectively (P = 0.16). In men, there was a significant decrease in BMDTBSDS during 3 years of GH, while BMAD LSSDS remained similar. In women, both BMDTBSDS and BMADLSSDS remained similar. BMDTBSDS was associated with female sex, lean body mass and age. The majority of patients received sex steroid replacement therapy (SSRT). Conclusions: During 3 years of combined GH and SSRT treatment, BMD remained stable in the normal range in young adults with PWS. However, men showed a decline in BMD TBSDS, probably due to insufficient SSRT. We recommended to continue GH treatment in young adults with PWS a nd to start SSRT during adolescence unless puberty progresses normally.

AB - Objective: In children with Prader-Willi syndrome (PWS), growth hormone ( GH) treatment has positive effects on bone mineral density (BMD). Two 1-year studies did not show a differe nce between GH or placebo on BMD in young adults with PWS. However, there are no studies investigating BM D during longer-term GH treatment in young adults with PWS. Design: Open-label, a prospective study in 43 young adults with PWS. Methods: BMD of the total body (BMDTBSDS) and lumbar spine (BMADLSSDS) measured by DXA. Results: In the total group, estimated mean (95% CI) of BMD TB remained similar during 3 years of GH, being -0.76 (-1.11 to -0.41) SDS at start and -0.90 (-1.27 to -0.54) SDS after 3 years (P = 0.11), as did BMADLS, being -0.36 (-0.72 to 0.01) SDS and -0.46 (-0.77 to -0.16) SDS, respectively (P = 0.16). In men, there was a significant decrease in BMDTBSDS during 3 years of GH, while BMAD LSSDS remained similar. In women, both BMDTBSDS and BMADLSSDS remained similar. BMDTBSDS was associated with female sex, lean body mass and age. The majority of patients received sex steroid replacement therapy (SSRT). Conclusions: During 3 years of combined GH and SSRT treatment, BMD remained stable in the normal range in young adults with PWS. However, men showed a decline in BMD TBSDS, probably due to insufficient SSRT. We recommended to continue GH treatment in young adults with PWS a nd to start SSRT during adolescence unless puberty progresses normally.

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U2 - 10.1530/eje-20-1335

DO - 10.1530/eje-20-1335

M3 - Article

C2 - 33769952

AN - SCOPUS:85105537027

SN - 0804-4643

VL - 184

SP - 773

EP - 782

JO - European Journal of Endocrinology

JF - European Journal of Endocrinology

IS - 6

ER -

Bone mineral density during 3 years of growth hormone in previously GH-treated young adults with PWS

Abstract

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