{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T20:05:31Z","timestamp":1790193931163,"version":"4.1.0"},"reference-count":18,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2017,10,16]],"date-time":"2017-10-16T00:00:00Z","timestamp":1508112000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Clinical Endocrinology"],"published-print":{"date-parts":[[2017,12]]},"abstract":"<jats:title>Summary<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>Thymic neuroendocrine tumour (<jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content>) accounts for almost 20% of multiple endocrine neoplasia type 1 (<jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1)\u2010associated mortality. Identifying risk factors for the development of these rare tumours and prognostic factors for clinical outcomes will be helpful in clinical practice.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design and Patients<\/jats:title><jats:p>We performed a retrospective analysis of patients treated for <jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content> associated with <jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1 in a single institution and meta\u2010analysis of literature reports. We used a fixed effect model to pool results across studies to evaluate the prevalence, clinical features and prognosis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p><jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content> was detected in 9 (7.4%) of 121 patients with <jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1 seen in our institution, and 5 (55.6%) were women. Seven additional studies were identified through a systematic review of the literature. The pool estimate of <jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content> prevalence was 3.7% (n\u00a0=\u00a099) in <jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1 (n\u00a0=\u00a02710), sex ratio was 79:20 (male vs female), and the median age at diagnosis was 43.0\u00a0years (range, 16.0\u201072.0\u00a0years). Forty\u2010three patients died with a median survival time of 8.4\u00a0years. Older age at diagnosis (<jats:styled-content style=\"fixed-case\">HR<\/jats:styled-content>\u00a0=\u00a01.4, 95% <jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content>\u00a0=\u00a01.0\u20101.8, <jats:italic>P<\/jats:italic>\u00a0=\u00a0.03), maximum tumour diameter (<jats:styled-content style=\"fixed-case\">HR<\/jats:styled-content>\u00a0=\u00a01.5, 95% <jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content>\u00a0=\u00a01.0\u20102.3, <jats:italic>P<\/jats:italic>\u00a0=\u00a0.04) and presence of metastasis (<jats:styled-content style=\"fixed-case\">HR<\/jats:styled-content>\u00a0=\u00a01.6, 95% <jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content>\u00a0=\u00a01.0\u20102.5, <jats:italic>P<\/jats:italic>\u00a0=\u00a0.04) were associated with worse outcome. A male predominance (91.9% vs 59.5%, <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.001) and history of smoking (59.0% vs 23.5%, <jats:italic>P<\/jats:italic>\u00a0=\u00a0.015) were more common in American\/European series compared to Asian reports.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p><jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content> is a rare but fatal component of <jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1. Earlier detection of <jats:styled-content style=\"fixed-case\">TH<\/jats:styled-content>\u2010<jats:styled-content style=\"fixed-case\">NET<\/jats:styled-content> in patients with <jats:styled-content style=\"fixed-case\">MEN<\/jats:styled-content>1 may be recommended which should theoretically result in better outcomes. Different genetic backgrounds (race) appear to result in clinical difference.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cen.13480","type":"journal-article","created":{"date-parts":[[2017,9,20]],"date-time":"2017-09-20T09:12:48Z","timestamp":1505898768000},"page":"706-716","source":"Crossref","is-referenced-by-count":35,"title":["Clinical features and prognosis of thymic neuroendocrine tumours associated with multiple endocrine neoplasia type 1: A single\u2010centre study, systematic review and meta\u2010analysis"],"prefix":"10.1111","volume":"87","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4064-4460","authenticated-orcid":false,"given":"Lei","family":"Ye","sequence":"first","affiliation":[{"name":"Shanghai Key Laboratory for Endocrine Tumors Shanghai Clinical Center for Endocrine and Metabolic Diseases Shanghai Institute of Endocrine and Metabolic Diseases and Shanghai E\u2010institute for Endocrinology Ruijin Hospital Shanghai Jiaotong University, School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weixi","family":"Wang","sequence":"additional","affiliation":[{"name":"Shanghai Key Laboratory for Endocrine Tumors Shanghai Clinical Center for Endocrine and Metabolic Diseases Shanghai Institute of Endocrine and Metabolic Diseases and Shanghai E\u2010institute for Endocrinology Ruijin Hospital Shanghai Jiaotong University, School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naykky Singh","family":"Ospina","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Diabetes, Metabolism and Nutrition Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lei","family":"Jiang","sequence":"additional","affiliation":[{"name":"Shanghai Key Laboratory for Endocrine Tumors Shanghai Clinical Center for Endocrine and Metabolic Diseases Shanghai Institute of Endocrine and Metabolic Diseases and Shanghai E\u2010institute for Endocrinology Ruijin Hospital Shanghai Jiaotong University, School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ioannis","family":"Christakis","sequence":"additional","affiliation":[{"name":"Department of Surgical Oncology The University of Texas MD Anderson Cancer Center  Houston TX USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jieli","family":"Lu","sequence":"additional","affiliation":[{"name":"Shanghai Key Laboratory for Endocrine Tumors Shanghai Clinical Center for Endocrine and Metabolic Diseases Shanghai Institute of Endocrine and Metabolic Diseases and Shanghai E\u2010institute for Endocrinology Ruijin Hospital Shanghai Jiaotong University, School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yulin","family":"Zhou","sequence":"additional","affiliation":[{"name":"Shanghai Key Laboratory for Endocrine Tumors Shanghai Clinical Center for Endocrine and Metabolic 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