{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,23]],"date-time":"2026-06-23T16:56:36Z","timestamp":1782233796545,"version":"3.54.5"},"reference-count":20,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2026,4,5]],"date-time":"2026-04-05T00:00:00Z","timestamp":1775347200000},"content-version":"vor","delay-in-days":4,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T00:00:00Z","timestamp":1775001600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pathology International"],"published-print":{"date-parts":[[2026,4]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Oncocytic adrenal neoplasms are rare adrenocortical tumors characterized by abundant eosinophilic cytoplasm due to massive mitochondrial accumulation. Their biological behavior is often difficult to predict, and various diagnostic systems\u2014including the Lin\u2013Weiss\u2013Bisceglia system, the Helsinki score, and the reticulin algorithm\u2014are used to assess their malignant potential. We report a case of an oncocytic adrenocortical carcinoma associated with Cushing's syndrome and hyperandrogenemia in a 34\u2010year\u2010old woman. Histologically, the tumor showed diffuse growth of eosinophilic cells with marked pleomorphism and focal capsular invasion. Immunohistochemistry confirmed adrenocortical origin and cortisol production, with a Ki\u201067 labeling index of 25%. Ultrastructural examination revealed densely packed mitochondria with lamellar and tubulovesicular cristae, accompanied by numerous whorled smooth endoplasmic reticulum formations, suggesting active remodeling of the endoplasmic reticulum. Whole\u2010exome sequencing identified a pathogenic\n                    <jats:italic>GNAS<\/jats:italic>\n                    R201S mutation, together with copy number losses of\n                    <jats:italic>ARID1A<\/jats:italic>\n                    ,\n                    <jats:italic>CDKN2A<\/jats:italic>\n                    , and\n                    <jats:italic>ZNRF3<\/jats:italic>\n                    with widespread copy number alteration. Despite multiple adverse prognostic indicators, the patient has remained disease\u2010free for over 5 years following adrenalectomy and adjuvant low\u2010dose mitotane therapy. These findings suggest that\n                    <jats:italic>GNAS<\/jats:italic>\n                    activation may drive steroidogenesis while attenuating malignant progression, as demonstrated by integrated morphologic and genomic assessment in this case.\n                  <\/jats:p>","DOI":"10.1111\/pin.70112","type":"journal-article","created":{"date-parts":[[2026,4,5]],"date-time":"2026-04-05T16:10:23Z","timestamp":1775405423000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Cortisol\u2010Producing Oncocytic Adrenocortical Carcinoma Harboring a\n                    <i>GNAS<\/i>\n                    Mutation: An Integrated Histologic, Ultrastructural, and Genomic Analysis"],"prefix":"10.1111","volume":"76","author":[{"given":"Naomi","family":"Sato","sequence":"first","affiliation":[{"name":"Division of Pathological Diagnosis Tohoku Medical and Pharmaceutical University Sendai Miyagi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuta","family":"Tezuka","sequence":"additional","affiliation":[{"name":"Department of Diabetes, Metabolism, and Endocrinology Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taito","family":"Itoh","sequence":"additional","affiliation":[{"name":"Department of Investigative Pathology Tohoku University Graduate School of Medicine Sendai Miyagi Japan"},{"name":"Laboratory of Molecular Diagnostics and Therapeutics, Center for Intractable Diseases and ImmunoGenomics National Institutes of Biomedical Innovation, Health and Nutrition Ibaraki Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuko","family":"Omori","sequence":"additional","affiliation":[{"name":"Department of Investigative Pathology Tohoku University Graduate School of Medicine Sendai Miyagi 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Tohoku University Hospital Sendai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masaaki","family":"Kitada","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Faculty of Medicine Kansai Medical University Hirakata Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9976-2632","authenticated-orcid":false,"given":"Yuto","family":"Yamazaki","sequence":"additional","affiliation":[{"name":"Department of Pathology Tohoku University Hospital Sendai Miyagi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toru","family":"Furukawa","sequence":"additional","affiliation":[{"name":"Department of Investigative Pathology Tohoku University Graduate School of Medicine Sendai Miyagi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takashi","family":"Suzuki","sequence":"additional","affiliation":[{"name":"Department of Pathology Tohoku University Hospital Sendai Miyagi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6709-2876","authenticated-orcid":false,"given":"Yasuhiro","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Division of Pathology, Faculty of Medicine Tohoku Medical and Pharmaceutical University Sendai Miyagi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,4,5]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12022-014-9337-4"},{"key":"e_1_2_11_3_1","volume-title":"Endocrine and Neuroendocrine Tumours","author":"WHO Classification of Tumours Editorial 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