Evidence map›Paper›PMID 33815285›Full record

ArticleFrontiers in endocrinology2021

Quality of Life in Men With Congenital Adrenal Hyperplasia Due to 21-Hydroxylase Deficiency.

Myrthe J M Verhees, Manon Engels, Paul N Span, Fred C G J Sweep, Antonius E van Herwaarden, Henrik Falhammar, Anna Nordenström, Emma A Webb, Annette Richter-Unruh, Claire Bouvattier and 8 more

Open access · goldAbstract read
In one paragraph

Article in Frontiers in endocrinology, 2021. 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
0.7field-weighted citation impact, top 32% 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, 12 citations in OpenAlex.

  1. Guideline
  2. Article
  3. Article
  4. Clinical practice guidelines for the management of differences of sex development in Japan.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2026
    Review
  5. Article
  6. Review
  7. Article
  8. Article
  9. Long-Term Outcomes of Congenital Adrenal Hyperplasia.Endocrinology and metabolism (Seoul, Korea) · 2022
    Review
  10. Article
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

18 authors at 12 institutions in 5 countries.

Myrthe J M VerheesDepartment of Pediatrics, Amalia Children's Hospital, Radboud University Medical Center, Nijmegen, Netherlands.
Manon EngelsDepartment of Pediatrics, Amalia Children's Hospital, Radboud University Medical Center, Nijmegen, Netherlands.
Paul N SpanRadiotherapy and OncoImmunology Laboratory, Department of Radiation Oncology, Radboud Institute for Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, Netherlands.
Fred C G J SweepDepartment of Laboratory Medicine, Radboud Institute for Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, Netherlands.
Antonius E van HerwaardenDepartment of Laboratory Medicine, Radboud Institute for Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, Netherlands.
Henrik FalhammarDepartment of Molecular Medicine and Surgery, Karolinska Institute, Stockholm, Sweden.
Anna NordenströmDepartment of Women's and Children's Health, Division of Pediatric Endocrinology, Karolinska Institutet, Karolinska University Hospital, Stockholm, Sweden.
Emma A WebbCentre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, United Kingdom.
Annette Richter-UnruhSektion Kinderendokrinologie und Diabetologie, Klinik für Kinder- und Jugendmedizin der Ruhr-Universität Bochum im St. Josef-Hospital, Bochum, Germany.
Claire BouvattierEndocrinologie Pédiatrique, Centre de Référence des Maladies Rares du Développement Sexuel, Hôpital Bicêtre, Université Paris-Sud, Le Kremlin-Bicêtre, France.
Aude Brac de la PerrièreFédération d'Endocrinologie, Centre de Référence des Maladies Rares du Développement Génital, Groupement Hospitalier Est, Hopital Louis Pradel, Bron, France.
Wiebke ArltCentre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, United Kingdom.
Nicole ReischMedizinische Klinik IV, Klinikum der Universität München, München, Germany.
Birgit KöhlerKlinik für Pädiatrie m.S. Endokrinologie und Diabetologie, Charité - Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin, and Berlin Institute of Health, Berlin, Germany.
Marion RappKlinik fur Kinder- und Jugendmedizin, Universitat zu Lubeck, Lubeck, Germany.
Nike M M L StikkelbroeckDepartment of Internal Medicine, Division of Endocrinology, Radboud University Medical Center, Nijmegen, Netherlands.
Nel RoeleveldDepartment for Health Evidence, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, Netherlands.
Hedi L Claahsen-van der GrintenDepartment of Pediatrics, Amalia Children's Hospital, Radboud University Medical Center, Nijmegen, Netherlands.
Radboud University Medical Center · NLRadboud Institute for Molecular Life Sciences · NLRadboud University Nijmegen · NLUniversity of Birmingham · GBFreie Universität Berlin · DEHôpital Louis Pradel · FRKarolinska Institutet · SEKarolinska University Hospital · SELMU Klinikum · DESt. Josef-Hospital · DEUniversité Paris-Sud · FRUniversity of Lübeck · DE

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Congenital adrenal hyperplasia (CAH) due to 21-hydroxylase deficiency (21OHD) is a disorder of adrenal steroid biosynthesis, leading to hypocortisolism, hypoaldosteronism, and hyperandrogenism. Impaired quality of life (QoL) has been demonstrated in women with CAH, but data on men with CAH are scarce. We hypothesized that disease severity and poor treatment control are inversely associated with QoL. In this study, 109 men (16-68 years) with 21OHD were included. The WHOQOL-BREF questionnaire was used to measure self-reported QoL domain scores on a 0-100 scale, where higher scores reflect better QoL. QoL domain scores were compared to published data on healthy and chronically ill reference populations from France, Germany, the Netherlands, and the United Kingdom. Differences in QoL scores among groups of disease severity and treatment control were tested within the study population. Overall, the men with CAH in this study appeared to rate their QoL as good. Median domain scores were 78.6 (IQR: 67.9-85.7) for physical health, 79.2 (IQR: 66.7-87.5) for psychological health, 75.0 (IQR: 58.3-83.3) for social relationships, and 81.3 (IQR: 71.9-90.6) for environment. In general, these scores were similar to WHOQOL-BREF domain scores in healthy references and higher compared to chronically ill reference populations. The domain scores did not differ among genotype groups, but patients with undertreatment or increased 17-hydroxyprogestrone concentrations scored higher on several QoL domains (p<0.05). Patients treated with dexamethasone or prednisone scored higher on the physical health, psychological health, and social relationships domains, but not on the environmental domain. In conclusion, QoL domain scores appeared to be comparable to healthy reference populations and higher compared to patients with a chronic illness. QoL was not influenced by genotype, but undertreatment and use of dexamethasone or prednisone were associated with higher QoL.

Indexed as

Mental HealthAdrenal Hyperplasia, CongenitalAdultHumansMaleQuality of LifeSelf ReportSeverity of Illness IndexSurveys and QuestionnairesYoung Adult21 hydroxylase deficiencycongenital adrenal hyperplasia (CAH)CYP21A2quality of lifeWHOQOL BREF

Identifiers

PMID33815285
PMCPMC8018222
OpenAlexW3137225802

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.