Evidence map›Paper›PMID 34640379›Full record

ArticleJournal of clinical medicine2021

Hypogonadism in Adult Males with Prader-Willi Syndrome-Clinical Recommendations Based on a Dutch Cohort Study, Review of the Literature and an International Expert Panel Discussion.

Karlijn Pellikaan, Yassine Ben Brahim, Anna G W Rosenberg, Kirsten Davidse, Christine Poitou, Muriel Coupaye, Anthony P Goldstone, Charlotte Høybye, Tania P Markovic, Graziano Grugni and 10 more

Open access · goldAbstract read
In one paragraph

Article in Journal of clinical medicine, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed, 1 pooled it
3.8field-weighted citation impact, top 6% 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

15 citing papers in PubMed, 1 synthesis or guideline pooled it, 27 citations in OpenAlex.

  1. Pooled it
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  3. GH Therapy in Non-Growth Hormone-Deficient Children.Children (Basel, Switzerland) · 2024
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

20 authors at 12 institutions in 9 countries.

Karlijn PellikaanDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.ORCID 0000-0002-0738-0275
Yassine Ben BrahimDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.
Anna G W RosenbergDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.ORCID 0000-0002-8333-1512
Kirsten DavidseDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.
Christine PoitouAssistance Publique-Hôpitaux de Paris, Rare Diseases Center of Reference 'Prader-Willi Syndrome and Obesity with Eating Disorders' (PRADORT), Nutrition Department, Pitié-Salpêtrière Hospital, F-75013 Paris, France.
Muriel CoupayeAssistance Publique-Hôpitaux de Paris, Rare Diseases Center of Reference 'Prader-Willi Syndrome and Obesity with Eating Disorders' (PRADORT), Nutrition Department, Pitié-Salpêtrière Hospital, F-75013 Paris, France.ORCID 0000-0002-8988-6260
Anthony P GoldstoneInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).ORCID 0000-0001-8179-7071
Charlotte HøybyeInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).ORCID 0000-0002-3980-1927
Tania P MarkovicInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).ORCID 0000-0003-2521-0357
Graziano GrugniInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).ORCID 0000-0003-4708-2544
Antonino CrinòInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).
Assumpta CaixàsInternational Network for Research, Management & Education on Adults with PWS (INfoRMEd-PWS).ORCID 0000-0001-8472-9189
Talia Eldar-GevaThe Israel Multidisciplinary Prader-Willi Syndrome Clinic, Jerusalem 9103102, Israel.
Harry J HirschThe Israel Multidisciplinary Prader-Willi Syndrome Clinic, Jerusalem 9103102, Israel.ORCID 0000-0002-7499-4023
Varda Gross-TsurThe Israel Multidisciplinary Prader-Willi Syndrome Clinic, Jerusalem 9103102, Israel.
Merlin G ButlerDepartments of Psychiatry, Behavioral Sciences and Pediatrics, University of Kansas Medical Center, Kansas City, KS 66160, USA.ORCID 0000-0002-2911-0524
Jennifer L MillerDepartment of Pediatrics, College of Medicine, University of Florida, Gainesville, FL 32610, USA.
Sjoerd A A van den BergDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.
Aart J van der LelyDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.ORCID 0000-0002-1059-0126
Laura C G de GraaffDepartment of Internal Medicine, Division of Endocrinology, Erasmus University Medical Center Rotterdam, 3015 GD Rotterdam, The Netherlands.
Erasmus MC · NLHebrew University of Jerusalem · ILSorbonne Université · FRBambino Gesù Children's Hospital · ITImperial College Healthcare NHS Trust · GBIRCCS Istituto Auxologico Italiano · ITKarolinska University Hospital · SEShaare Zedek Medical Center · ILThe University of Sydney · AUUniversitat Autònoma de Barcelona · ESUniversity of Florida · USUniversity of Kansas Medical Center · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Prader-Willi syndrome (PWS) is a complex genetic syndrome characterized by hyperphagia, intellectual disability, hypotonia and hypothalamic dysfunction. Adults with PWS often have hormone deficiencies, hypogonadism being the most common. Untreated male hypogonadism can aggravate PWS-related health issues including muscle weakness, obesity, osteoporosis, and fatigue. Therefore, timely diagnosis and treatment of male hypogonadism is important. In this article, we share our experience with hypogonadism and its treatment in adult males with PWS and present a review of the literature. In order to report the prevalence and type of hypogonadism, treatment regimen and behavioral issues, we retrospectively collected data on medical interviews, physical examinations, biochemical measurements and testosterone replacement therapy (TRT) in 57 Dutch men with PWS. Fifty-six (98%) of the patients had either primary, central or combined hypogonadism. Untreated hypogonadism was associated with higher body mass index and lower hemoglobin concentrations. TRT was complicated by behavioral challenges in one third of the patients. Undertreatment was common and normal serum testosterone levels were achieved in only 30% of the patients. Based on the Dutch cohort data, review of the literature and an international expert panel discussion, we provide a practical algorithm for TRT in adult males with PWS in order to prevent undertreatment and related adverse health outcomes.

Indexed as

hypogonadismobesitypituitary glandPrader-Willi syndromepubertytestosterone

Identifiers

PMID34640379
PMCPMC8509256
OpenAlexW3202865650

What OpenQuestion holds

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LicenceCC BY
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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.