Evidence map›Paper›PMID 30973645›Full record

ArticleClinical endocrinology2019

Prevalence of growth hormone (GH) deficiency in previously GH-treated young adults with Prader-Willi syndrome.

Stephany H Donze, Layla Damen, Janiëlle A E M van Alfen-van der Velden, Gianni Bocca, Martijn J J Finken, Gera J G Hoorweg-Nijman, Petr E Jira, Mariëtte van Leeuwen, Anita C S Hokken-Koelega

Open access · hybridAbstract read
In one paragraph

Article in Clinical endocrinology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed, 1 pooled it
3.2field-weighted citation impact, top 8% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

10 citing papers in PubMed, 1 synthesis or guideline pooled it, 23 citations in OpenAlex.

  1. Growth Hormone Treatment for Adults With Prader-Willi Syndrome: A Meta-Analysis.The Journal of clinical endocrinology and metabolism · 2021
    Pooled it
  2. Review
  3. Article
  4. Diagnosis and management of sleep disorders in Prader-Willi syndrome.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2022
    Review
  5. Article
  6. Article
  7. Article
  8. Review
  9. Central Adrenal Insufficiency Is Rare in Adults With Prader-Willi Syndrome.The Journal of clinical endocrinology and metabolism · 2020
    Article
  10. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

9 authors at 7 institutions in 1 country.

Stephany H DonzeDutch Growth Research Foundation, Rotterdam, The Netherlands.ORCID 0000-0002-9249-4284
Layla DamenDutch Growth Research Foundation, Rotterdam, The Netherlands.
Janiëlle A E M van Alfen-van der VeldenDepartment of Pediatrics, Subdivision of Endocrinology, Radboud University Medical Centre, Amalia Children's Hospital, Nijmegen, The Netherlands.
Gianni BoccaDepartment of Pediatrics, Subdivision of Endocrinology, University Medical Center Groningen, Beatrix Children's Hospital, Groningen, The Netherlands.
Martijn J J FinkenDepartment of Pediatrics, Subdivision of Endocrinology, VU University Medical Center, Amsterdam, The Netherlands.ORCID 0000-0002-6589-9788
Gera J G Hoorweg-NijmanDepartment of Pediatrics, St. Antonius Hospital, Utrecht, The Netherlands.
Petr E JiraDepartment of Pediatrics, Jeroen Bosch Hospital, 's-Hertogenbosch, The Netherlands.
Mariëtte van LeeuwenDepartment of Pediatrics, St. Jansdal, Harderwijk, The Netherlands.
Anita C S Hokken-KoelegaDutch Growth Research Foundation, Rotterdam, The Netherlands.
Erasmus MC · NLAmsterdam UMC Location Vrije Universiteit Amsterdam · NLJan de Rijk Logistics (Netherlands) · NLJeroen Bosch Ziekenhuis · NLRadboud University Nijmegen · NLSt. Antonius Ziekenhuis · NLUniversity Medical Center Groningen · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveSome features of subjects with Prader-Willi syndrome (PWS) resemble those seen in growth hormone deficiency (GHD). Children with PWS are treated with growth hormone (GH), which has substantially changed their phenotype. Currently, young adults with PWS must discontinue GH after attainment of adult height when they do not fulfil the criteria of adult GHD. Limited information is available about the prevalence of GHD in adults with PWS. This study aimed to investigate the GH/insulin-like growth factor (IGF-I) axis and the prevalence of GHD in previously GH-treated young adults with PWS.

designCross-sectional study in 60 young adults with PWS. MEASUREMENTS: Serum IGF-I and IGFBP-3 levels, GH peak during combined growth hormone-releasing hormone (GHRH)-arginine stimulation test.

resultsSerum IGF-I was <-2 standard deviation scores (SDS) in 2 (3%) patients, and IGFBP-3 was within the normal range in all but one patient. Median (IQR) GH peak was 17.8 μg/L (12.2; 29.7) [~53.4 mU/L] and below 9 μg/L in 9 (15%) patients. Not one patient fulfilled the criteria for adult GHD (GH peak < 9 μg/L and IGF-I < -2 SDS), also when BMI-dependent criteria were used. A higher BMI and a higher fat mass percentage were significantly associated with a lower GH peak. There was no significant difference in GH peak between patients with a deletion or a maternal uniparental disomy (mUPD).

conclusionsIn a large group of previously GH-treated young adults with PWS, approximately 1 in 7 exhibited a GH peak <9 μg/L during a GHRH-arginine test. However, none of the patients fulfilled the consensus criteria for adult GHD.

Indexed as

AdultBody Mass IndexCross-Sectional StudiesDwarfism, PituitaryFemaleGrowth HormoneHuman Growth HormoneHumansInsulin-Like Growth Factor Binding Protein 3Insulin-Like Growth Factor IMalePrader-Willi SyndromePrevalenceYoung AdultGrowth HormoneHuman Growth HormoneIGFBP3 protein, humanInsulin-Like Growth Factor Binding Protein 3Insulin-Like Growth Factor Iargininegrowth hormonegrowth hormone deficiencygrowth hormone-releasing hormoneIGF-IPrader-Willi syndrome

Identifiers

PMID30973645
PMCPMC6850120
OpenAlexW2938011265

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.