{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T00:50:27Z","timestamp":1790297427964,"version":"4.1.0"},"reference-count":12,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,8,12]],"date-time":"2021-08-12T00:00:00Z","timestamp":1628726400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,8,12]],"date-time":"2021-08-12T00:00:00Z","timestamp":1628726400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Endocr Disord"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Various adrenal disorders including primary aldosteronism and Cushing\u2019s syndrome lead to the cause of hypertension. Although primary aldosteronism is sometimes complicated with preclinical Cushing\u2019s syndrome, concurrence of overt Cushing\u2019s syndrome and primary aldosteronism is very rare. In addition, it has been drawing attention recently that primary aldosteronism is brought about by the presence of aldosterone-producing cell cluster in adjacent adrenal cortex rather than the presence of aldosterone-producing adenoma.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 67-year-old Japanese female was referred to our institution due to moon face and central obesity. Based on various clinical findings and data, we diagnosed this subject as overt Cushing\u2019s syndrome and primary aldosteronism. Furthermore, in immunostaining for cytochrome P450 (CYP) 11B1, a cortisol-producing enzyme, diffuse staining was observed in tumorous lesion. Also, in immunostaining for CYP11B2, an aldosterone-producing enzyme, CYP11B2 expression was not observed in tumorous lesion, but strong CYP11B2 expression was observed in adjacent adrenal cortex, indicating the presence of aldosterone-producing cell cluster.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>We should bear in mind the possibility that concurrence of overt Cushing\u2019s syndrome and primary aldosteronism is accompanied by aldosterone-producing cell cluster in adjacent adrenal cortex.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-021-00818-2","type":"journal-article","created":{"date-parts":[[2021,8,12]],"date-time":"2021-08-12T09:03:03Z","timestamp":1628758983000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Concurrence of overt Cushing\u2019s syndrome and primary aldosteronism accompanied by aldosterone-producing cell cluster in adjacent adrenal cortex: case report"],"prefix":"10.1186","volume":"21","author":[{"given":"Yoshiro","family":"Fushimi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fuminori","family":"Tatsumi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junpei","family":"Sanada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masashi","family":"Shimoda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinji","family":"Kamei","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shuhei","family":"Nakanishi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kohei","family":"Kaku","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomoatsu","family":"Mune","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7898-1943","authenticated-orcid":false,"given":"Hideaki","family":"Kaneto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,8,12]]},"reference":[{"key":"818_CR1","doi-asserted-by":"publisher","first-page":"331","DOI":"10.1161\/HYPERTENSIONAHA.113.01060","volume":"62","author":"E Savard","year":"2013","unstructured":"Savard E, Amar L, Plouin PF, Steichen O. Cardiovascular complications associated with primary aldosteronism: a controlled cross-sectional study. Hypertension. 2013;62:331\u20136.","journal-title":"Hypertension"},{"key":"818_CR2","doi-asserted-by":"publisher","first-page":"723","DOI":"10.1291\/hypres.24.723","volume":"24","author":"T Honda","year":"2001","unstructured":"Honda T, Nakamura T, Saito Y, Ohyama Y, Sumino H, Kurabayashi M. Combined primary aldosteronism and preclinical Cushing\u2019s syndrome: an unusual case presentation of adrenal adenoma. Hypertens Res. 2001;24:723\u201336.","journal-title":"Hypertens Res"},{"key":"818_CR3","doi-asserted-by":"publisher","first-page":"714","DOI":"10.2169\/internalmedicine.42.714","volume":"42","author":"J Adachi","year":"2003","unstructured":"Adachi J, Hirai Y, Terui K, Nakao T, Fukuda Y, Suda T, Sasano H. A report of 7 cases of adrenal tumors secreting both cortisol and aldosterone. Intern Med. 2003;42:714\u20138.","journal-title":"Intern Med"},{"issue":"1425","key":"818_CR4","first-page":"1427","volume":"69","author":"H Fujii","year":"2005","unstructured":"Fujii H, Kamide K, Miyake O, Abe T, Nagai M, Nakahama H, Horio T, Takiuchi S, Okuyama A, Yutani C, Kawano Y. Primary aldosteronism combined with preclinical Cushing\u2019s syndrome in an elderly patient. Circ J. 2005;69(1425):1427.","journal-title":"Circ J"},{"key":"818_CR5","doi-asserted-by":"publisher","first-page":"242","DOI":"10.1097\/MED.0000000000000248","volume":"23","author":"CE Gomez-Sanchez","year":"2016","unstructured":"Gomez-Sanchez CE, Gomez-Sanchez EP. Immunohistochemistry of the adrenal in primary aldosteronism. Curr Opin Endocrinol Diabetes Obes. 2016;23:242\u20138.","journal-title":"Curr Opin Endocrinol Diabetes Obes"},{"key":"818_CR6","doi-asserted-by":"publisher","first-page":"7834356","DOI":"10.1210\/jc.2015-3285","volume":"2016","author":"K Nishimoto","year":"2016","unstructured":"Nishimoto K, Seki T, Hayashi Y, Mikami S, Al-Eyd G, Nakagawa K, Morita S, Kosaka T, Oya M, Mitani F, Suematsu M, Kabe Y, Mukai K. Human adrenocortical remodeling leading to aldosterone-producing cell cluster generation. Int J Endocrinol. 2016;2016:7834356. https:\/\/doi.org\/10.1210\/jc.2015-3285.","journal-title":"Int J Endocrinol"},{"key":"818_CR7","doi-asserted-by":"publisher","first-page":"6","DOI":"10.1210\/jc.2015-3285","volume":"101","author":"K Nishimoto","year":"2016","unstructured":"Nishimoto K, Seki T, Kurihara I, Yokota K, Omura M, Nishikawa T, Shibata H, Kosaka T, Oya M, Suematsu M, Mukai K. Case report: nodule development from subcapsular aldosterone-producing cell clusters causes hyperaldosteronism. J Clin Endocrinol Metab. 2016;101:6\u20139.","journal-title":"J Clin Endocrinol Metab"},{"key":"818_CR8","doi-asserted-by":"publisher","first-page":"951","DOI":"10.1055\/s-0043-122394","volume":"49","author":"K Omata","year":"2017","unstructured":"Omata K, Tomlins SA, Rainey WE. Aldosterone-producing cell clusters in normal and pathological states. Horm Metab Res. 2017;49:951\u20136.","journal-title":"Horm Metab Res"},{"key":"818_CR9","doi-asserted-by":"publisher","first-page":"427","DOI":"10.1055\/a-1128-0421","volume":"52","author":"JS Lim","year":"2020","unstructured":"Lim JS, Rainey WE. The potential role of aldosterone-producing cell clusters in adrenal disease. Horm Metab Res. 2020;52:427\u201334.","journal-title":"Horm Metab Res"},{"key":"818_CR10","doi-asserted-by":"publisher","first-page":"634","DOI":"10.1161\/HYPERTENSIONAHA.119.14041","volume":"75","author":"N Sun","year":"2020","unstructured":"Sun N, Meyer LS, Feuchtinger A, Kunzke T, Kn\u00f6sel T, Reincke M, Walch A, Williams TA. Mass spectrometry imaging establishes 2 distinct metabolic phenotypes of aldosterone-producing cell cClusters in primary aldosteronism. Hypertension. 2020;75:634\u201344.","journal-title":"Hypertension"},{"key":"818_CR11","doi-asserted-by":"publisher","first-page":"2712","DOI":"10.1210\/jc.2013-4146","volume":"99","author":"V Lim","year":"2014","unstructured":"Lim V, Guo Q, Grant CS, Thompson GB, Richards ML, Farley DR, Yong WF Jr. Accuracy of adrenal imaging and adrenal venous sampling in predicting surgical cure of primary aldosteronism. J Clin Endocrinol Metab. 2014;99:2712\u20139.","journal-title":"J Clin Endocrinol Metab"},{"key":"818_CR12","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1161\/HYPERTENSIONAHA.113.02097","volume":"63","author":"GP Rossi","year":"2014","unstructured":"Rossi GP, Auchus RJ, Brown M, Lenders JWM, Naruse M, Plouin PF, Satoh F, Yong WF Jr. An expert consensus statement on use of adrenal vein sampling for the subtyping of primary aldosteronism. Hypertension. 2014;63:151\u201360.","journal-title":"Hypertension"}],"container-title":["BMC Endocrine Disorders"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-021-00818-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12902-021-00818-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12902-021-00818-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,8,12]],"date-time":"2021-08-12T09:04:02Z","timestamp":1628759042000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcendocrdisord.biomedcentral.com\/articles\/10.1186\/s12902-021-00818-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,8,12]]},"references-count":12,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["818"],"URL":"https:\/\/doi.org\/10.1186\/s12902-021-00818-2","relation":{},"ISSN":["1472-6823"],"issn-type":[{"value":"1472-6823","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,8,12]]},"assertion":[{"value":"25 March 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"18 July 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"12 August 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"We do not have any potential conflicts of interest relevant to this article.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"163"}}