Evidence map›Paper›PMID 41779164›Full record

ArticleThe Journal of clinical endocrinology and metabolism2026

Health-related quality of life in children with multiple endocrine neoplasia (MEN) and their siblings.

Daniël Zwerus, Annemarie A Verrijn Stuart, Hanneke M van Santen, Gerlof D Valk, Christiaan de Bruin, Theo C J Sas, Gianni Bocca, Christiaan F Mooij, Hedi L Claahsen-van der Grinten, Saartje Straetemans and 2 more

Abstract read
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Article in The Journal of clinical endocrinology and metabolism, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

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2citing papers in PubMed
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1 · What the graph read from it

What it found

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2 · The registry

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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

2 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

12 authors.

Daniël ZwerusDepartment of Endocrine Oncology, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands.ORCID 0000-0002-0545-8569
Annemarie A Verrijn StuartWilhelmina Children's Hospital, University Medical Center Utrecht, 3584 EA Utrecht, The Netherlands.
Hanneke M van SantenWilhelmina Children's Hospital, University Medical Center Utrecht, 3584 EA Utrecht, The Netherlands.
Gerlof D ValkDepartment of Endocrine Oncology, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands.ORCID 0000-0001-5841-8344
Christiaan de BruinWillem-Alexander Children's Hospital, LUMC, 2333 ZC Leiden, The Netherlands.ORCID 0000-0003-1478-2261
Theo C J SasDepartment of Pediatric Endocrinology, Erasmus Medical Center-Sophia's Children's Hospital, 3015 AA Rotterdam, The Netherlands.
Gianni BoccaBeatrix Children's Hospital, Department of Pediatrics, University Medical Center Groningen, University of Groningen, 9713 GZ Groningen, The Netherlands.ORCID 0000-0002-2665-8738
Christiaan F MooijDepartment of Pediatric Endocrinology, Emma's Children's Hospital, Amsterdam University Medical Center, University of Amsterdam and Vrije Universiteit, 1105 AZ Amsterdam, The Netherlands.ORCID 0000-0001-7977-6271
Hedi L Claahsen-van der GrintenDepartment of Pediatrics, Amalia's Children's Hospital, Radboud UMC, 6525 GA Nijmegen, The Netherlands.
Saartje StraetemansMosaKids Children's Hospital, Maastricht UMC, 6229 HX Maastricht, The Netherlands.ORCID 0000-0001-7857-3608
Sasja A SchepersWilhelmina Children's Hospital, University Medical Center Utrecht, 3584 EA Utrecht, The Netherlands.
Rachel S van LeeuwaardeDepartment of Endocrine Oncology, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands.ORCID 0000-0001-6147-9746

Funding

Children Cancer Free Foundation
6 · The paper itself

Abstract

contextMultiple Endocrine Neoplasia (MEN) syndromes are rare autosomal dominant hereditary tumor predisposition syndromes affecting multiple family members. Carriers undergo health surveillance from early childhood onwards. Carriership or surveillance may influence Health-Related Quality of Life (HRQoL) for patients or their families.

objectiveTo evaluate HRQoL in children and adolescents with genetically confirmed MEN1, MEN2A, and MEN2B, and to compare outcomes with their siblings without MEN and healthy Dutch norms. Secondary aims were to explore associations between HRQoL and clinical characteristics.

methodsThis nationwide cross-sectional study included 77 children with MEN (5-18 years) and 26 siblings (8-29 years). The Pediatric Quality of Life Inventory (PedsQL) was used to assess HRQoL, with children completing self-report questionnaires and parents providing proxy reports for their children with MEN. Sociodemographic and clinical data were obtained from medical records.

resultsChildren with MEN1 and MEN2A reported HRQoL comparable to siblings and healthy Dutch norms. Only children with MEN2B showed significantly lower physical HRQoL. No gender or age effects were observed. Parent-proxy scores were significantly higher than child self-reports on all domains, except for emotional functioning. Children with MEN1 and MEN2B having clinical MEN-related manifestations had significantly lower physical, social, and school functioning scores than those without clinical manifestations.

conclusionOverall, children with MEN reported HRQoL comparable to siblings and age-matched Dutch norms, except for reduced physical functioning in MEN2B. Clinical manifestations negatively affected physical, social, and school functioning, suggesting that children with the onset of disease may benefit from closer monitoring and targeted psychosocial support.

Indexed as

Multiple Endocrine NeoplasiaMultiple Endocrine Neoplasia Type 1Quality of LifeSiblingsAdolescentAdultChildChild, PreschoolCross-Sectional StudiesFemaleHumansMaleNetherlandsSurveys and QuestionnairesYoung Adultchildrenmultiple endocrine neoplasiaparentspsychosocialquality of lifesiblings

Identifiers

PMID41779164
PMCPMC13368371

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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.