One Year of Recombinant Human Growth Hormone Treatment in Adults with Prader-Willi Syndrome Improves Body Composition, Motor Skills and Brain Functional Activity in the Cerebellum.
Prader–Willi syndrome
fMRI
growth hormone
hypotonia
motor function
myokines
Journal
Journal of clinical medicine
ISSN: 2077-0383
Titre abrégé: J Clin Med
Pays: Switzerland
ID NLM: 101606588
Informations de publication
Date de publication:
25 Mar 2022
25 Mar 2022
Historique:
received:
15
02
2022
revised:
18
03
2022
accepted:
24
03
2022
entrez:
12
4
2022
pubmed:
13
4
2022
medline:
13
4
2022
Statut:
epublish
Résumé
We compared body composition, biochemical parameters, motor function, and brain neural activation in 27 adults with Prader-Willi syndrome and growth-hormone deficiency versus age-and sex-matched controls and baseline versus posttreatment values of these parameters after one year of recombinant human growth hormone (rhGH) treatment. To study body composition, we analyzed percentage of fat mass, percentage of lean mass, and muscle-mass surrogate variables from dual X-ray absorptiometry. Biochemical parameters analyzed included IGF-I, glucose metabolism, and myokines (myostatin, irisin, and IL6). To explore muscle function, we used dynamometer-measured handgrip strength, the Timed Up and Go (TUG) test, and the Berg Balance Scale (BBS). To study brain activation, we acquired functional magnetic resonance images during three motor tasks of varying complexity. After one year of treatment, we observed an increase in lean mass and its surrogates, a decrease in fat mass, improvements in TUG test and BBS scores, and increased neural activation in certain cerebellar areas. The treatment did not significantly worsen glucose metabolism, and no side-effects were reported. Our findings support the benefits of rhGH treatment in adults with Prader-Willi syndrome and growth-hormone deficiency on body composition and suggest that it may also improve balance and brain neural activation.
Identifiants
pubmed: 35407437
pii: jcm11071831
doi: 10.3390/jcm11071831
pmc: PMC8999376
pii:
doi:
Types de publication
Journal Article
Langues
eng
Subventions
Organisme : Instituto de Salud Carlos III
ID : PI-18/00734
Organisme : Institute of Research and Innovation Parc Tauli
ID : CIR 2020/022
Références
J Int Med Res. 2018 Nov;46(11):4669-4677
pubmed: 30213215
Nature. 1997 May 1;387(6628):83-90
pubmed: 9139826
J Appl Physiol (1985). 2007 Sep;103(3):1093-8
pubmed: 17347387
BMC Musculoskelet Disord. 2009 May 06;10:47
pubmed: 19419559
Res Dev Disabil. 2011 Jan-Feb;32(1):81-6
pubmed: 20884170
Sci Rep. 2020 Jan 23;10(1):1020
pubmed: 31974460
Neuroimage. 2014 Jan 1;84:320-41
pubmed: 23994314
Neuroimage. 2014 Nov 1;101:87-95
pubmed: 24999036
Neuroscience. 2011 Apr 14;179:131-42
pubmed: 21277357
J Clin Sleep Med. 2017 May 15;13(5):665-666
pubmed: 28416048
Md State Med J. 1965 Feb;14:61-5
pubmed: 14258950
J Clin Med. 2021 Jun 17;10(12):
pubmed: 34204309
Growth Horm IGF Res. 2000 Apr;10 Suppl B:S87-91
pubmed: 10984260
Nature. 2012 Jan 11;481(7382):463-8
pubmed: 22237023
J Clin Med. 2021 Jul 28;10(15):
pubmed: 34362104
Curr Probl Pediatr. 1984 Jan;14(1):1-55
pubmed: 6365470
Neurosci Biobehav Rev. 2012 Sep;36(8):1817-38
pubmed: 22652271
Endocr Rev. 2001 Dec;22(6):787-99
pubmed: 11739333
J Am Geriatr Soc. 1991 Feb;39(2):142-8
pubmed: 1991946
J Clin Endocrinol Metab. 2014 May;99(5):1816-24
pubmed: 24471571
J Intellect Disabil Res. 2005 Apr;49(Pt 4):245-52
pubmed: 15816811
Acta Paediatr. 2006 Jul;95(7):861-3
pubmed: 16801186
Neurosci Biobehav Rev. 2011 Jan;35(3):956-69
pubmed: 21056055
Acta Paediatr Suppl. 1997 Nov;423:50-4
pubmed: 9401539
Am J Med Genet A. 2020 Jan;182(1):115-121
pubmed: 31692257
J Neuroeng Rehabil. 2007 May 10;4:14
pubmed: 17493259
Physiol Rev. 2008 Oct;88(4):1379-406
pubmed: 18923185
Am J Med Genet A. 2007 Mar 1;143A(5):443-8
pubmed: 17103437
J Child Neurol. 2007 Jan;22(1):38-40
pubmed: 17608303
PLoS One. 2015 Sep 03;10(9):e0136864
pubmed: 26334732
J Gerontol. 1994 Mar;49(2):M85-94
pubmed: 8126356
Actas Esp Psiquiatr. 2019 May;47(3):79-87
pubmed: 31233206
Metabolism. 2012 Dec;61(12):1725-38
pubmed: 23018146
Can J Public Health. 1992 Jul-Aug;83 Suppl 2:S7-11
pubmed: 1468055
Growth Horm IGF Res. 2011 Aug;21(4):185-90
pubmed: 21664161
Clin Endocrinol (Oxf). 2012 Jul;77(1):86-93
pubmed: 22117629
Arch Phys Med Rehabil. 1970 Jun;51(6):321-7
pubmed: 5423802
Handb Clin Neurol. 2018;154:59-70
pubmed: 29903452
Clin Endocrinol (Oxf). 2021 Apr;94(4):645-655
pubmed: 33296095
J Endocrinol Invest. 2008 Sep;31(9):765-72
pubmed: 18997487
J Nucl Med. 2006 Jul;47(7):1088-92
pubmed: 16818941
PeerJ. 2017 Dec 13;5:e4097
pubmed: 29255649
Cerebellum. 2020 Dec;19(6):778-787
pubmed: 32661798
Endocrinol Diabetes Nutr (Engl Ed). 2019 Nov;66(9):579-587
pubmed: 31006652
Psychoneuroendocrinology. 2019 May;103:67-75
pubmed: 30658340
Med Sci Sports Exerc. 2011 Oct;43(10):1828-35
pubmed: 21364474
Genet Med. 2012 Jan;14(1):10-26
pubmed: 22237428
Int J Mol Sci. 2018 Jan 26;19(2):
pubmed: 29373545
Endocrine. 2012 Apr;41(2):191-9
pubmed: 22081257
Arch Neurol. 2004 Oct;61(10):1585-9
pubmed: 15477513
J Neurol Sci. 1969 Jul-Aug;9(1):49-61
pubmed: 5820859
J Endocrinol Invest. 2008 Feb;31(2):169-75
pubmed: 18362510
Dev Neuropsychol. 2009;34(3):272-83
pubmed: 19437203
J Pediatr Endocrinol Metab. 2004 Sep;17(9):1153-75
pubmed: 15506675
Endocr Pract. 2016 Oct;22(10):1235-1244
pubmed: 27409821
J Int Neuropsychol Soc. 2021 Nov;27(10):1024-1036
pubmed: 33660593
J Endocrinol Invest. 2021 Oct;44(10):2261-2271
pubmed: 33656700
J Neurosurg. 1998 May;88(5):863-9
pubmed: 9576255
Brain Dev. 1994 May-Jun;16(3):183-8
pubmed: 7943601
Adv Pediatr. 2016 Aug;63(1):47-77
pubmed: 27426895
Disabil Rehabil. 2009;31(21):1780-4
pubmed: 19479512
PLoS One. 2013 Apr 11;8(4):e60563
pubmed: 23593248
Growth Horm IGF Res. 2013 Jun;23(3):81-7
pubmed: 23433655
Am J Ment Retard. 2004 Mar;109(2):142-53
pubmed: 15000675