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We describe an MCMT case presented in a 56-year-old woman with a history of arterial hypertension and high levels of aldosterone, accompanied by a review of the literature. The adrenalectomy specimen showed a well-circumscribed nodule of 30\u00a0mm in size, containing 60% of cells with a cortical phenotype (positive for \u03b1-inhibin and melan-A) and 40% of cells with a medullary phenotype (positive for chromogranin-A, GATA-3 and somatostatin receptor 2). There was no significant mitotic activity, necrosis, nor lymphovascular invasion. The\n                    <jats:italic>GNAS<\/jats:italic>\n                    p.(Arg844Cys) mutation, as well as variants of uncertain significance\n                    <jats:italic>AKAP13<\/jats:italic>\n                    p.(His641Pro) and\n                    <jats:italic>EPAS1<\/jats:italic>\n                    p.(Ser478del) were detected in the tumor. MCMT is more common in women (75%) with a mean age of 46.6\u00a0years (range 16\u201378). Most patients present with hypertension (79%), frequently associated with Cushing\u2019s syndrome, (39%), diabetes (21%), aldosteronism (15%), and\/or hyperandrogenism (6%). Laboratory data showed elevated levels of both cortisol and cathecholamines and\/or their metabolites in more than 50% of cases, supporting the dual nature of the tumor. Most MCMTs are benign, but aggressive behavior was detected in four (12%) cases, all of them showing large size (80\u2013220\u00a0mm), poor delimitation, venous invasion, necrosis, and\/or high proliferation rates. The pathogenesis is unknown, but our findings suggest a tumor histogenesis from the cortical cellular component through the regulation of the protein kinase A pathway and secondary proliferation of the medullary component.\n                  <\/jats:p>","DOI":"10.1007\/s00428-025-04091-9","type":"journal-article","created":{"date-parts":[[2025,4,4]],"date-time":"2025-04-04T09:03:07Z","timestamp":1743757387000},"page":"367-375","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Adrenal mixed corticomedullary tumors: report of a case with molecular characterization and systematic review"],"prefix":"10.1007","volume":"488","author":[{"ORCID":"https:\/\/orcid.org\/0009-0008-7589-6012","authenticated-orcid":false,"given":"Edurne","family":"P\u00e9rez-B\u00e9liz","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9117-2646","authenticated-orcid":false,"given":"Benjam\u00edn Alfonso","family":"Thorpe-Plaza","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5202-638X","authenticated-orcid":false,"given":"Everardo Josu\u00e9","family":"D\u00edaz-L\u00f3pez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-6684-8315","authenticated-orcid":false,"given":"Lourdes","family":"Loidi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-6920-0577","authenticated-orcid":false,"given":"Carmen","family":"Villalba-Mart\u00edn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1247-2331","authenticated-orcid":false,"given":"Ihab","family":"Abdulkader-Nallib","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5516-8914","authenticated-orcid":false,"given":"Jos\u00e9 Manuel","family":"Cameselle-Teijeiro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,4,2]]},"reference":[{"issue":"4","key":"4091_CR1","doi-asserted-by":"publisher","first-page":"635","DOI":"10.1016\/0002-9343(69)90193-4","volume":"47","author":"DA Mathison","year":"1969","unstructured":"Mathison DA, Waterhouse CA (1969) Cushing\u2019s syndrome with hypertensive crisis and mixed adrenal cortical adenoma-pheochromocytoma (corticomedullary adenoma). 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