{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T03:09:41Z","timestamp":1787022581890,"version":"build-2736575974"},"reference-count":35,"publisher":"Walter de Gruyter GmbH","issue":"6","license":[{"start":{"date-parts":[[2019,5,31]],"date-time":"2019-05-31T00:00:00Z","timestamp":1559260800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,6,26]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec id=\"j_jpem-2018-0444_s_999_w2aab3b7c17b1b6b1aab1c16b1Aa\">\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>In Japan, prophylactic thyroidectomy involves out-of-pocket expense. The American Thyroid Association (ATA) recommends prophylactic thyroidectomy for medullary thyroid carcinoma (MTC) during early childhood in patients with multiple endocrine neoplasia type 2 (MEN2). The ATA reports a high frequency of postoperative complications in childhood, which also influenced the delay of prophylactic thyroidectomy in Japan.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"j_jpem-2018-0444_s_998_w2aab3b7c17b1b6b1aab1c16b2Aa\">\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      This retrospective study of multiple medical centers in Japan included individuals aged &lt;20 years diagnosed with germline\n                      <jats:italic>RET<\/jats:italic>\n                      mutations between 1997 and 2017. The onset and onset possibility were defined based on confirmed lesions or calcitonin levels. The definition of risk and prophylactic thyroidectomy were based on the ATA 2015 revised guideline.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"j_jpem-2018-0444_s_997_w2aab3b7c17b1b6b1aab1c16b3Aa\">\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Twenty-one patients with MEN2 were enrolled (highest risk, n\u2009=\u20095; high risk, n\u2009=\u20095; and moderate risk, n\u2009=\u200911). The cumulative incidence of the onset\/onset possibility reached 50% at 5 and 8 years and 100% at 9 years and 17 years in high- and moderate-risk patients, respectively. Of 7 patients with MEN2A, 71% underwent prophylactic thyroidectomy. Only one 5-year-old patient (C634Y) had increased serum calcitonin level after prophylactic thyroidectomy in the MEN2A group. The only permanent complication, which did not occur in patients who underwent total thyroidectomy alone, was hypoparathyroidism (33% of patients). This permanent complication occurred with clinically developed MTC. No permanent postoperative complications occurred in patients aged 5\u20136 years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"j_jpem-2018-0444_s_996_w2aab3b7c17b1b6b1aab1c16b4Aa\">\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Prophylactic thyroidectomy reduces recurrence and postoperative complications in pediatric patients with MEN2. Early thyroidectomy based on only calcitonin level could possibly reduce thyroidectomy delay.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1515\/jpem-2018-0444","type":"journal-article","created":{"date-parts":[[2019,5,31]],"date-time":"2019-05-31T05:03:15Z","timestamp":1559278995000},"page":"585-595","source":"Crossref","is-referenced-by-count":11,"title":["Present status of prophylactic thyroidectomy in pediatric multiple endocrine neoplasia 2: a nationwide survey in Japan 1997\u20132017"],"prefix":"10.1515","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3328-9499","authenticated-orcid":false,"given":"Rie","family":"Matsushita","sequence":"first","affiliation":[{"name":"Division of Pediatrics , Kikugawa General Hospital , Shizuoka , Japan"},{"name":"Department of Pediatrics , Hamamatsu University School of Medicine , Shizuoka , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keisuke","family":"Nagasaki","sequence":"additional","affiliation":[{"name":"Division of Pediatrics, Department of Homeostatic Regulation and Development , Niigata University Graduate School of Medical and Dental Sciences , Niigata , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tadayuki","family":"Ayabe","sequence":"additional","affiliation":[{"name":"Japan Environment and Children\u2019s Study Medical Support Center , National Research Institute for Child Health and Development , Tokyo , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoko","family":"Miyoshi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics , Osaka University Graduate School of Medicine , Osaka , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saori","family":"Kinjo","sequence":"additional","affiliation":[{"name":"Division of Pediatrics , Okinawa Chubu Hospital , Okinawa , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hidenori","family":"Haruna","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Adolescent Medicine , Juntendo University Graduate School of Medicine , Tokyo , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenji","family":"Ihara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine , Oita University , Oita , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomonobu","family":"Hasegawa","sequence":"additional","affiliation":[{"name":"Department of Pediatrics , Keio University School of Medicine , Tokyo , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinobu","family":"Ida","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Endocrinology , Osaka Women\u2019s and Children\u2019s Hospital , Osaka , Japan"},{"name":"Pediatric Chronic Intestinal Pseudo-Obstruction (CIPO), Research Group for \u201cComprehensive Study and Seamless Guidelines\u201d on Rare and Intractable Gastrointestinal Disease from Childhood, Supported by the Ministry of Health, Labor and Welfare of Japan , Tokyo , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keiichi","family":"Ozono","sequence":"additional","affiliation":[{"name":"Department of Pediatrics , Osaka University Graduate School of Medicine , Osaka , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kanshi","family":"Minamitani","sequence":"additional","affiliation":[{"name":"Department of Pediatrics , Teikyo University Chiba Medical Center , 3426-3 Anesaki, Ichihara-shi , Chiba 299-0111 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2019,5,31]]},"reference":[{"key":"2023040101024194467_j_jpem-2018-0444_ref_001_w2aab3b7c17b1b6b1ab2b2b1Aa","doi-asserted-by":"crossref","unstructured":"Machens A, Dralle H. 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