{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T00:46:43Z","timestamp":1787014003827,"version":"build-2736575974"},"reference-count":30,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2022,4,22]],"date-time":"2022-04-22T00:00:00Z","timestamp":1650585600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Endocrinology"],"published-print":{"date-parts":[[2022,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>We recently reported cases of adipsic hypernatremia caused by autoantibodies against the subfornical organ in patients with hypothalamic\u2010pituitary lesions. This study aimed to clarify the clinical features of newly identified patients with adipsic hypernatremia whose sera displayed immunoreactivity to the mouse subfornical organ.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>Observational cohort study of patients diagnosed with adipsic hypernatremia in Japan, United States, and Europe.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The study included 22 patients with adipsic hypernatremia but without overt structural changes in the hypothalamic\u2010pituitary region and congenital disease. Antibody response to the mouse subfornical organ was determined using immunohistochemistry. The clinical characteristics were compared between the patients with positive and negative antibody responses.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Antibody response to the mouse subfornical organ was detected in the sera of 16 patients (72.7%, female\/male ratio, 1:1, 12 pediatric and 4 adult patients). The prolactin levels at the time of diagnosis were significantly higher in patients with positive subfornical organ (SFO) immunoreactivity than in those with negative SFO immunoreactivity (58.9\u2009\u00b1\u200933.5 vs. 22.9\u2009\u00b1\u200913.9\u2009ng\/ml,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u2009.05). Hypothalamic disorders were found in 37.5% of the patients with positive SFO immunoreactivity. Moreover, six patients were diagnosed with rapid\u2010onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation\/neural tumor syndrome after the diagnosis of adipsic hypernatremia. Plasma renin activity levels were significantly higher in patients with serum immunoreactivity to the Na\n                      <jats:sub>x<\/jats:sub>\n                      channel.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The patients with serum immunoreactivity to the SFO had higher prolactin levels and hypothalamic disorders compared to those without the immunoreactivity. The clinical characteristics of patients with serum immunoreactivity to the subfornical organ included higher prolactin levels and hypothalamic disorders, which were frequently associated with central hypothyroidism and the presence of retroperitoneal tumors.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/cen.14737","type":"journal-article","created":{"date-parts":[[2022,4,14]],"date-time":"2022-04-14T01:53:14Z","timestamp":1649901194000},"page":"72-80","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Identification of clinical factors related to antibody\u2010mediated immune response to the subfornical organ"],"prefix":"10.1111","volume":"97","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7417-8923","authenticated-orcid":false,"given":"Akari","family":"Nakamura\u2010Utsunomiya","sequence":"first","affiliation":[{"name":"Department of Pediatrics Hiroshima University Graduate School of Biomedical and Health Sciences Hiroshima Japan"},{"name":"Department of Pediatrics Hiroshima Prefectural Hospital Hiroshima Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoshi","family":"Goda","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Hiroshima University Graduate School of Biomedical and Health Sciences Hiroshima Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seiichi","family":"Hayakawa","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Hiroshima University Hospital Hiroshima Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sakata","family":"Sonoko","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Hiroshima University Graduate School of Biomedical and Health Sciences Hiroshima Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8738-3571","authenticated-orcid":false,"given":"Ewout J.","family":"Hoorn","sequence":"additional","affiliation":[{"name":"Division of Nephrology and Transplantation, Department of Internal Medicine Erasmus MC, University Medical Center Rotterdam Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0815-0586","authenticated-orcid":false,"given":"Anne","family":"Blanchard","sequence":"additional","affiliation":[{"name":"Center of Clinical investigation Faculty of Medicine, Hospital European George Pompidou Paris Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akiko","family":"Saito\u2010Hakoda","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haruna","family":"Kakimoto","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Kagoshima University Hospital Kagoshima Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8412-3494","authenticated-orcid":false,"given":"Rumi","family":"Hachiya","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism Tokyo Metropolitan Children's Medical Center Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miki","family":"Kamimura","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rie","family":"Kawakita","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Metabolism Children's Medical Center, Osaka City General Hospital Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinji","family":"Higuchi","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Metabolism Children's Medical Center, Osaka City General Hospital Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rika","family":"Fujimaru","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Osaka City General Hospital Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3734-4766","authenticated-orcid":false,"given":"Yoko","family":"Shirai","sequence":"additional","affiliation":[{"name":"Department of Pediatric Nephrology Tokyo Women's Medical University Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daichi","family":"Miyaoka","sequence":"additional","affiliation":[{"name":"Department of Metabolism, Endocrinology and Molecular Medicine Osaka City University Graduate School of Medicine Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuki","family":"Nagata","sequence":"additional","affiliation":[{"name":"Department of Vascular Medicine Osaka City University Graduate School of Medicine Osaka Japan"},{"name":"Department of vascular medicine Vascular Science Center for Translational Research, Osaka City University Graduate School of Medicine Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yutaro","family":"Kishi","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism Niigata University Medical and Dentist Hospital Niigata Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aya","family":"Wada","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Gunma University Hospital Tochigi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akari","family":"Mitsuboshi","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism Hyogo Prefectural Kobe children's Hospital Kobe Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kayo","family":"Ozaki","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism Hyogo Prefectural Kobe children's Hospital Kobe Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nagisa","family":"Komatsu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Kumamoto Chuo Hospital Kumamoto Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3161-8968","authenticated-orcid":false,"given":"Hidetaka","family":"Niizuma","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junko","family":"Kanno","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3622-4375","authenticated-orcid":false,"given":"Ikuma","family":"Fujiwara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1288-9455","authenticated-orcid":false,"given":"Yukihiro","family":"Hasegawa","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism Tokyo Metropolitan Children's Medical Center Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8945-4775","authenticated-orcid":false,"given":"Tohru","family":"Yorifuji","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Metabolism Children's Medical Center, Osaka City General Hospital Osaka 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