{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2022,8,29]],"date-time":"2022-08-29T08:12:09Z","timestamp":1661760729034},"reference-count":61,"publisher":"The Endocrine Society","issue":"8","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[1999,8,1]]},"abstract":"<jats:p>Negative sellar exploration (despite the results of endocrine evaluation indicating Cushing\u2019s disease), the high incidence of failure of total hypophysectomy, and remission of Cushing\u2019s syndrome after unsuccessful hypophysectomy and sellar irradiation suggest that the etiology of refractory Cushing\u2019s disease, in some patients, lies near the sella but not in the pituitary gland. We present 5 patients, out of 626 who received surgery for Cushing\u2019s disease, in whom an ACTH-secreting extrapituitary parasellar adenoma was identified: 2 after unsuccessful total hypophysectomy for the treatment of refractory Cushing\u2019s disease, 2 after unsuccessful hemihypophysectomy (the first, 2 yr before treatment at the NIH for Nelson\u2019s syndrome; and the second, with recurrent Cushing\u2019s disease 5 yr after negative transsphenoidal exploration), and 1 with a preoperative diagnosis of an intraclival microadenoma, which was cured by resection of the tumor. In all cases, an extrapituitary parasellar microadenoma was confirmed unequivocally as the cause of the disease, by negative pathology of the resected pituitary gland (patients 1, 2, 3, and 5), and\/or the remission of the disease after selective resection of the extrasellar adenoma (patients 3, 4, and 5). Three of 5 patients had a partial empty sella. These patients support the thesis that ACTH-secreting tumors can arise exclusively from remnants of Rathke\u2019s pouch, rather than from the adenohypophysis (anterior lobe or pars tuberalis of the pituitary gland) and can be a cause of Cushing\u2019s disease. In the sixth presented case, an extrapituitary tumor was suspected at surgery after negative pituitary exploration, but serial sections of the hemihypophysectomy specimen revealed a microscopic focus of tumor at the margin of the resected gland. This case demonstrates the importance of negative pituitary histology to establish the presence of an extrapituitary parasellar tumor as an exclusive source of ACTH, and it supports the value of clinical outcome to establish the diagnosis with selective adenomectomy of an extrapituitary parasellar tumor. In patients with negative pituitary magnetic resonance imaging, especially in the presence of a partial empty sella, the diagnostic and surgical approach in Cushing\u2019s disease should consider the identification and resection of extrapituitary parasellar adenoma, which can avoid total hypophysectomy, as was possible in 3 of our 5 patients.<\/jats:p>","DOI":"10.1210\/jcem.84.8.5890","type":"journal-article","created":{"date-parts":[[2014,1,8]],"date-time":"2014-01-08T17:04:31Z","timestamp":1389200671000},"page":"2912-2923","source":"Crossref","is-referenced-by-count":6,"title":["Extrapituitary Parasellar Microadenoma in Cushing\u2019s Disease"],"prefix":"10.1210","volume":"84","author":[{"given":"Ryszard M.","family":"Pluta","sequence":"first","affiliation":[{"name":"Surgical Neurology Branch (R.M.P., J.C.W., E.H.O.), Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lynnette","family":"Nieman","sequence":"additional","affiliation":[{"name":"National Institute of Neurological Disorders and Stroke, and Developmental Endocrinology Branch (L.N.), Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"John L.","family":"Doppman","sequence":"additional","affiliation":[{"name":"National Institute of Child Health and Human Development, Radiology Department (J.L.D.), Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Joseph C.","family":"Watson","sequence":"additional","affiliation":[{"name":"Surgical Neurology Branch (R.M.P., J.C.W., E.H.O.), Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nancy","family":"Tresser","sequence":"additional","affiliation":[{"name":"Clinical Center, Office of the Clinical Director (N.T., D.A.K.), NINDS, and Laboratory of Pathology, NCI, National Institutes of Health, Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David A.","family":"Katz","sequence":"additional","affiliation":[{"name":"Clinical Center, Office of the Clinical Director (N.T., D.A.K.), NINDS, and Laboratory of Pathology, NCI, National Institutes of Health, Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Edward H.","family":"Oldfield","sequence":"additional","affiliation":[{"name":"Surgical Neurology Branch (R.M.P., J.C.W., E.H.O.), Bethesda, Maryland 20892"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"80","reference":[{"key":"2020071618160870700_R1"},{"key":"2020071618160870700_R2","doi-asserted-by":"crossref","first-page":"897","DOI":"10.1056\/NEJM199109263251301","article-title":"Petrosal sinus sampling with and without corticotropin-releasing hormone for the differential diagnosis of Cushing\u2019s syndrome[ published erratum appears in N Engl J Med 1992 Apr 23;326(17):1172] [see comments].","volume":"325","author":"Oldfield","year":"1991","journal-title":"N Engl J Med"},{"key":"2020071618160870700_R3","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1056\/NEJM197107292850501","article-title":"Results of treatment in 108 patients with Cushing\u2019s syndrome.","volume":"285","author":"Orth","year":"1971","journal-title":"N Engl J Med"},{"key":"2020071618160870700_R4","doi-asserted-by":"crossref","first-page":"363","DOI":"10.3171\/jns.1990.72.3.0363","article-title":"Cushing\u2019s disease: results of transsphenoidal microsurgery with emphasis on surgical failures.","volume":"72","author":"Tindall","year":"1990","journal-title":"J Neurosurg"},{"key":"2020071618160870700_R5","doi-asserted-by":"crossref","first-page":"925","DOI":"10.1016\/S0889-8529(18)30075-6","article-title":"Cushing\u2019s disease. 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An aid in the evaluation of patients with Cushing\u2019s syndrome.","volume":"310","author":"Chrousos","year":"1984","journal-title":"N Engl J Med"},{"key":"2020071618160870700_R34","doi-asserted-by":"crossref","first-page":"647","DOI":"10.7326\/0003-4819-94-5-647","article-title":"Selective venous sampling for ACTH in Cushing\u2019s syndrome: differentiation between Cushing\u2019s disease and the ectopic ACTH syndrome.","volume":"94","author":"Findling","year":"1981","journal-title":"Ann Intern Med"},{"key":"2020071618160870700_R35","first-page":"1308","article-title":"A simplified morning ovine corticotrophin-releasing hormone stimulation test for differential diagnosis of ACTH-dependent Cushing\u2019s syndrome.","volume":"77","author":"Nieman","year":"1993","journal-title":"J Clin Endocrinol Metab"},{"key":"2020071618160870700_R36","doi-asserted-by":"crossref","first-page":"418","DOI":"10.1210\/jcem.78.2.8106630","article-title":"A comparison of the standard high-dose dexamethasone suppression test for the differential diagnosis of Cushing syndrome.","volume":"78","author":"Dichek","year":"1994","journal-title":"J Clin Endocrinol Metab"},{"key":"2020071618160870700_R37","doi-asserted-by":"crossref","first-page":"587","DOI":"10.1056\/NEJM198502283120921","article-title":"Cushing\u2019s disease caused by an ectopic pituitary adenoma within the sphenoid sinus.","volume":"312","author":"Burch","year":"1985","journal-title":"N Engl J Med"},{"key":"2020071618160870700_R38"},{"key":"2020071618160870700_R39","first-page":"138","article-title":"ACTH and TSH producing ectopic suprasellar pituitary adenoma of the hypothalamic region: case report.","volume":"12","author":"Lindboe","year":"1993","journal-title":"Clin Neuropathol"},{"key":"2020071618160870700_R40","doi-asserted-by":"crossref","first-page":"267","DOI":"10.1007\/BF00735033","article-title":"Ectopic anterior pituitary corticotropic tumour in a six-year-old boy. 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