Early start of growth hormone is associated with positive effects on auxology and metabolism in Prader-Willi-syndrome.

Child Developmental Biology Growth Hormone Humans Longitudinal Studies Male Prader-Willi Syndrome / drug therapy Retrospective Studies
Carbohydrate and lipid metabolism Growth hormone therapy Insulin-like growth factor-I Prader-Willi-syndrome

Journal

Orphanet journal of rare diseases
ISSN: 1750-1172
Titre abrégé: Orphanet J Rare Dis
Pays: England
ID NLM: 101266602

Informations de publication

Date de publication:
12 10 2020
Historique:
received: 16 05 2020
accepted: 07 09 2020
entrez: 13 10 2020
pubmed: 14 10 2020
medline: 22 6 2021
Statut: epublish

Résumé

Prader-Willi-Syndrome (PWS) is characterized by hypothalamic-pituitary dysfunction. Recent research suggests starting growth hormone-treatment (GHT) as soon as possible. The aim of this study is to analyze possible differences in auxological parameters, carbohydrate and lipid metabolism between two groups of children with PWS that started GHT either during or after their first year of life. Retrospective longitudinal study of 62 children (31 males) with genetically confirmed PWS. Upon diagnosis all children were offered GHT, some started immediately, others commenced later. Cohort A (n = 21; 11 males) started GHT at 0.3-0.99 yrs. (mean 0.72 yrs) and Cohort B (n = 41; 20 males) commenced GHT at 1.02-2.54 yrs. (mean 1.42 yrs) of age. Fasting morning blood samples and auxological parameters were obtained before the start of therapy and semi-annually thereafter. Differences between the two cohorts were estimated with a linear mixed-effect model. Mean length/height-SDS An early start of GHT during the first year of life seems to have a favorable effect on height-SDS and metabolic parameters.

Sections du résumé

BACKGROUND
Prader-Willi-Syndrome (PWS) is characterized by hypothalamic-pituitary dysfunction. Recent research suggests starting growth hormone-treatment (GHT) as soon as possible. The aim of this study is to analyze possible differences in auxological parameters, carbohydrate and lipid metabolism between two groups of children with PWS that started GHT either during or after their first year of life.
STUDY DESIGN
Retrospective longitudinal study of 62 children (31 males) with genetically confirmed PWS. Upon diagnosis all children were offered GHT, some started immediately, others commenced later. Cohort A (n = 21; 11 males) started GHT at 0.3-0.99 yrs. (mean 0.72 yrs) and Cohort B (n = 41; 20 males) commenced GHT at 1.02-2.54 yrs. (mean 1.42 yrs) of age. Fasting morning blood samples and auxological parameters were obtained before the start of therapy and semi-annually thereafter. Differences between the two cohorts were estimated with a linear mixed-effect model.
RESULTS
Mean length/height-SDS
CONCLUSIONS
An early start of GHT during the first year of life seems to have a favorable effect on height-SDS and metabolic parameters.

Identifiants

DOI: 10.1186/s13023-020-01527-0 PMID: 33046090 PMC: PMC7552493
pubmed: 33046090
doi: 10.1186/s13023-020-01527-0
pii: 10.1186/s13023-020-01527-0
pmc: PMC7552493
doi:

Substances chimiques

Growth Hormone 9002-72-6

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

283

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Auteurs

Lucy Magill (L)

Department of Pediatric Endocrinology and Diabetology, Children's Hospital, University of Bonn, Venusberg-Campus, Building 30, 53127, Bonn, Germany.

Constanze Laemmer (C)

Pediatric Endocrinology and Diabetology, St. Bernward Hospital, Treibestraße 9, 31134, Hildesheim, Germany.

Joachim Woelfle (J)

Children's University Hospital Erlangen, Loschgestrasse 15, 91054, Erlangen, Germany.

Rolf Fimmers (R)

University Hospital of Bonn, Institute for Medical Biometry, University of Bonn, Venusberg-Campus, 53127, Bonn, Germany.

Bettina Gohlke (B)

Department of Pediatric Endocrinology and Diabetology, Children's Hospital, University of Bonn, Venusberg-Campus, Building 30, 53127, Bonn, Germany. Bettina-gohlke@t-online.de.
ORCID: 0000-0003-1432-1465

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Classifications MeSH

questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Early start of growth hormone is associated...
questionsmedicales.fr › Disciplines des sciences naturelles › Disciplines des sciences biologiques › Biologie › Biologie du développement › Early start of growth hormone is associated...
questionsmedicales.fr › Acides aminés, peptides et protéines › Peptides › Hormones peptidiques › Hormones hypophysaires › Hormones antéhypophysaires › Hormone de croissance › Early start of growth hormone is associated...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Early start of growth hormone is associated...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Études longitudinales › Early start of growth hormone is associated...
questionsmedicales.fr › Maladies métaboliques et nutritionnelles › Troubles nutritionnels › Surnutrition › Syndrome de Prader-Willi › Early start of growth hormone is associated...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Études rétrospectives › Early start of growth hormone is associated...