{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T14:02:02Z","timestamp":1790172122211,"version":"4.1.0"},"reference-count":61,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T00:00:00Z","timestamp":1767571200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,1,5]],"date-time":"2026-01-05T00:00:00Z","timestamp":1767571200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"published-print":{"date-parts":[[2026,3]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>The combination of disease manifestations, the familial burden, and varying penetrance of endocrine tumor syndromes (ETSs) is unique. This review aimed to portray and summarize available data on psychosocial outcomes in patients with ETSs and explore gaps and opportunities for future research and care.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Literature was systematically searched for articles on psychosocial outcomes in patients with multiple endocrine neoplasia (MEN), von Hippel\u2013Lindau (VHL), and pathogenic variants (PVs) in succinate dehydrogenase (SDHx) genes via PubMed, PsychInfo, and Embase. Study quality was assessed using the CASP and STROBE appraisal tools.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>In total, 36 studies were found with fluctuating levels of evidence, of which five included pediatric patients. Overall, studies showed a considerable impact of ETSs on psychosocial outcomes such as quality of life (QoL), anxiety, and depression. Maladaptive coping, as well as social and financial restraints, were associated with poorer psychosocial outcomes. Surveillance protocols had ambivalent effects, both creating a sense of control and serving as a constant reminder of having an ETS. Parathyroid disease was associated with adverse psychosocial effects in MEN1. In MEN2A, gastrointestinal symptoms and having affected offspring were associated with poorer psychosocial outcomes. In MEN2B, pain was reported to interfere with daily life. Studies regarding VHL reported a wide range of experiences in patients and family members. Only a few studies were found for patients with PVs in SDHx genes, mainly describing effects due to the manifestation of disease or paraganglioma.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Psychosocial outcomes and possible underlying factors seem different for each ETS. More research is needed to address psychosocial outcomes in children with an ETS.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/1545-5017.70037","type":"journal-article","created":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T05:49:38Z","timestamp":1767678578000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Psychosocial Outcomes in Patients With Endocrine Tumor Syndromes: A Systematic Review"],"prefix":"10.1002","volume":"73","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0545-8569","authenticated-orcid":false,"given":"Dani\u00ebl","family":"Zwerus","sequence":"first","affiliation":[{"name":"Department of Endocrine Oncology University Medical Center Utrecht  Utrecht The Netherlands"},{"name":"Department of Psycho\u2010Oncology Princess M\u00e1xima Center for Pediatric Oncology  Utrecht The Netherlands"},{"name":"Department of Pediatric Endocrinology Wilhelmina Children's Hospital  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Floortje","family":"Strobbe","sequence":"additional","affiliation":[{"name":"Department of Endocrine Oncology University Medical Center Utrecht  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annemarie A.","family":"Verrijn Stuart","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology Wilhelmina Children's Hospital  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hanneke M.","family":"van Santen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology Wilhelmina Children's Hospital  Utrecht The Netherlands"},{"name":"Department of Supportive Care Princess M\u00e1xima Center for Pediatric Oncology  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gerlof D.","family":"Valk","sequence":"additional","affiliation":[{"name":"Department of Endocrine Oncology University Medical Center Utrecht  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sasja A.","family":"Schepers","sequence":"additional","affiliation":[{"name":"Department of Psycho\u2010Oncology Princess M\u00e1xima Center for Pediatric Oncology  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel S.","family":"van Leeuwaarde","sequence":"additional","affiliation":[{"name":"Department of Endocrine Oncology University Medical Center Utrecht  Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,1,5]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.3109\/08820538.2013.825281"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbabio.2011.07.003"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.physiol.62.1.377"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00520-016-3189-z"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm11195542"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11912-021-01029-7"},{"issue":"5","key":"e_1_2_9_8_1","first-page":"377","article-title":"Understanding the Increasing Incidence of Neuroendocrine Tumors","volume":"18","author":"Pathak S.","year":"2023","journal-title":"Expert Rev"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.seminoncol.2016.08.007"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.3322\/caac.21673"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1188\/06.ONF.221-231"},{"issue":"1","key":"e_1_2_9_12_1","first-page":"11","article-title":"Case Study of a Patient Family With Multiple Endocrine Neoplasia Syndrome. 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