Literature DB >> 9625332

Treatment with human growth hormone in patients with Prader-Labhart-Willi syndrome reduces body fat and increases muscle mass and physical performance.

U Eiholzer1, R Gisin, C Weinmann, S Kriemler, H Steinert, T Torresani, M Zachmann, A Prader.   

Abstract

UNLABELLED: Twelve children with documented Prader-Labhart-Willi syndrome were treated with human growth hormone (24 U/m2/week) during 1 year. The children were divided into three groups: group 1: overweight and prepubertal (n=6, age 3.8-7.0 years); group 2: underweight and prepubertal (n=3, age 0.6-4.1 years); group 3: pubertal (n=3, age 9.2-14.6 years). In group 1, height increased from -1.7 SD to -0.6 SD, while weight decreased from 1.1 SD to 0.4 SD, with a dramatic drop in weight for height from 3.8 SD to 1.2 SD. Hand length increased from -1.5 SD to -0.4 SD and foot length from -2.5 SD to -1.4 SD. Body fat, measured by dual X-ray energy absorptiometry, dropped by a third, whereas muscle mass increased by a fourth. Physical capability (Wingate test) improved considerably. The children were reported to be much more active and capable. In group 2, similar changes were seen, but weight for height increased, probably because muscle mass increase exceeded fat mass decrease. Changes in group 3 were similar as in group 1, even though far less distinct.
CONCLUSION: Growth hormone treatment in Prader-Labhart-Willi syndrome led to dramatic changes: distinct increase in growth velocity, height and muscle mass, as well as an improvement in physical performance. Fat mass and weight for height decreased in the initially overweight children, and weight for height increased in underweight children.

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Year:  1998        PMID: 9625332     DOI: 10.1007/s004310050832

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  24 in total

1.  Physical growth of Swiss children from birth to 20 years of age. First Zurich longitudinal study of growth and development.

Authors:  A Prader; R H Largo; L Molinari; C Issler
Journal:  Helv Paediatr Acta Suppl       Date:  1989-06

Review 2.  The Wingate anaerobic test. An update on methodology, reliability and validity.

Authors:  O Bar-Or
Journal:  Sports Med       Date:  1987 Nov-Dec       Impact factor: 11.136

3.  Molecular, cytogenetic, and clinical investigations of Prader-Willi syndrome patients.

Authors:  W P Robinson; A Bottani; Y G Xie; J Balakrishman; F Binkert; M Mächler; A Prader; A Schinzel
Journal:  Am J Hum Genet       Date:  1991-12       Impact factor: 11.025

4.  Growth hormone secretion and effects of growth hormone therapy on growth velocity and weight gain in children with Prader-Willi syndrome.

Authors:  M Angulo; M Castro-Magana; B Mazur; J A Canas; P M Vitollo; M Sarrantonio
Journal:  J Pediatr Endocrinol Metab       Date:  1996 May-Jun       Impact factor: 1.634

5.  Reduced growth hormone response to growth hormone-releasing hormone in children with simple obesity: evidence for somatomedin-C mediated inhibition.

Authors:  S Loche; M Cappa; P Borrelli; A Faedda; A Crinò; S G Cella; R Corda; E E Müller; C Pintor
Journal:  Clin Endocrinol (Oxf)       Date:  1987-08       Impact factor: 3.478

6.  Growth hormone (GH) response to combined pyridostigmine and GH-releasing hormone administration in patients with Prader-Labhard-Willi syndrome.

Authors:  M Cappa; A Grossi; P Borrelli; E Ghigo; J Bellone; S Benedetti; D Carta; S Loche
Journal:  Horm Res       Date:  1993

7.  Circulating somatomedin-C levels and the effect of growth hormone-releasing factor on plasma levels of growth hormone and somatostatin-like immunoreactivity in obese children.

Authors:  R Rosskamp; M Becker; S Soetadji
Journal:  Eur J Pediatr       Date:  1987-01       Impact factor: 3.183

8.  Magnetic resonance imaging evaluation of adipose tissue and muscle tissue mass in children with growth hormone (GH) deficiency, Turner's syndrome, and intrauterine growth retardation during the first year of treatment with GH.

Authors:  J Leger; C Carel; I Legrand; A Paulsen; M Hassan; P Czernichow
Journal:  J Clin Endocrinol Metab       Date:  1994-04       Impact factor: 5.958

9.  Body mass index in screening for adiposity in children and adolescents: systematic evaluation using receiver operating characteristic curves.

Authors:  R Lazarus; L Baur; K Webb; F Blyth
Journal:  Am J Clin Nutr       Date:  1996-04       Impact factor: 7.045

10.  Radioimmunological determination of insulinlike growth factors I and II in normal subjects and in patients with growth disorders and extrapancreatic tumor hypoglycemia.

Authors:  J Zapf; H Walter; E R Froesch
Journal:  J Clin Invest       Date:  1981-11       Impact factor: 14.808

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  15 in total

1.  Effects of growth hormone treatment in adults with Prader-Willi syndrome.

Authors:  M G Butler; B K Smith; J Lee; C Gibson; C Schmoll; W V Moore; J E Donnelly
Journal:  Growth Horm IGF Res       Date:  2013-02-19       Impact factor: 2.372

2.  Hyperphagia in male melanocortin 4 receptor deficient mice promotes growth independently of growth hormone.

Authors:  H Y Tan; F J Steyn; L Huang; M Cowley; J D Veldhuis; C Chen
Journal:  J Physiol       Date:  2016-10-02       Impact factor: 5.182

3.  Growth standards of infants with Prader-Willi syndrome.

Authors:  Merlin G Butler; Jennifer Sturich; Jaehoon Lee; Susan E Myers; Barbara Y Whitman; June-Anne Gold; Virginia Kimonis; Ann Scheimann; Norma Terrazas; Daniel J Driscoll
Journal:  Pediatrics       Date:  2011-03-14       Impact factor: 7.124

4.  Growth hormone treatment in a girl with Prader Willi syndrome.

Authors:  S N Pandey; R A Vaidya; A Irani
Journal:  Indian J Pediatr       Date:  2003-04       Impact factor: 1.967

5.  The snoRNA MBII-52 (SNORD 115) is processed into smaller RNAs and regulates alternative splicing.

Authors:  Shivendra Kishore; Amit Khanna; Zhaiyi Zhang; Jingyi Hui; Piotr J Balwierz; Mihaela Stefan; Carol Beach; Robert D Nicholls; Mihaela Zavolan; Stefan Stamm
Journal:  Hum Mol Genet       Date:  2010-01-06       Impact factor: 6.150

6.  Development and implementation of electronic growth charts for infants with Prader-Willi syndrome.

Authors:  S Trent Rosenbloom; Merlin G Butler
Journal:  Am J Med Genet A       Date:  2012-08-17       Impact factor: 2.802

7.  Clinical management of behavioral characteristics of Prader-Willi syndrome.

Authors:  Alan Y Ho; Anastasia Dimitropoulos
Journal:  Neuropsychiatr Dis Treat       Date:  2010-05-06       Impact factor: 2.570

8.  Long-term growth hormone therapy changes the natural history of body composition and motor function in children with prader-willi syndrome.

Authors:  Aaron L Carrel; Susan E Myers; Barbara Y Whitman; Jens Eickhoff; David B Allen
Journal:  J Clin Endocrinol Metab       Date:  2010-01-08       Impact factor: 5.958

9.  Growth Charts for Prader-Willi Syndrome During Growth Hormone Treatment.

Authors:  Merlin G Butler; Jaehoon Lee; Devin M Cox; Ann M Manzardo; June-Anne Gold; Jennifer L Miller; Elizabeth Roof; Elisabeth Dykens; Virginia Kimonis; Daniel J Driscoll
Journal:  Clin Pediatr (Phila)       Date:  2016-02-03       Impact factor: 1.168

Review 10.  Growth hormone treatment in children: review of safety and efficacy.

Authors:  Mark Harris; Paul L Hofman; Wayne S Cutfield
Journal:  Paediatr Drugs       Date:  2004       Impact factor: 3.022

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