Ectopic ACTH syndrome due to bilateral ovarian androblastoma with double, gynandroblastic differentiation in one ovary

Kasperlik-Załuska, A; Sikorowa, L; Ploch, E; Soszyński, P; Jeske, Wojciech; Migdalska, B; Wysocki, Maciej

doi:10.1016/0028-2243(93)90077-p

Cited by 12 publications

(3 citation statements)

References 13 publications

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“…Moreover, it is worth noting that immunohistochemistry may help in the differential diagnosis demonstrating immunoreactivities for neuroendocrine markers (chromogranin A and synaptophysin), generally lacking in ovarian adenocarcinomas, which are conversely immunoreactive for CEA and ER. The trabecular/acinar structure may also open the differential diagnosis with sex cord-stromal tumor of the ovary, which has also been described in association with Cushing's syndrome [23,24]. However, the lack of inhibin and ER staining and the immunoreactivity for general neuroendocrine markers help to solve this issue.…”

Section: Discussionmentioning

confidence: 91%

Cushing’s Syndrome due to a Pancreatic Neuroendocrine Tumor Metastatic to the Ovaries: a Clinicopathological Description of a Case

Rosa¹,

Marando

Ghezzi

et al. 2011

Endocr Pathol

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We report the case of a 36-year-old woman with Cushing's syndrome caused by a malignant unresectable neuroendocrine carcinoma of the pancreas that developed bilateral ovarian metastases 7 years after diagnosis. In November 2001, because of abdominal pain and jaundice, the patient underwent radiological investigations and exploratory laparotomy that demonstrated the presence of a 3-cm mass of the head of the pancreas, infiltrating the superior mesenteric vein, associated with enlargement of multiple abdominal lymph nodes and with a liver nodule. Histological examination of one lymph node and of the liver nodule demonstrated the presence of metastases from a well-differentiated neuroendocrine carcinoma showing corticotropin immunoreactivity. A few months later, the patient started to show the clinical symptoms of Cushing's syndrome and underwent steroid-blocking ketoconazole therapy. The clinical endocrine picture was controlled until the end of 2008, when the endocrine symptoms of the Cushing's syndrome worsened and bilateral ovarian tumors appeared. Hysteroannexectomy was performed and ovarian tumors were found to be metastases from a well-differentiated neuroendocrine carcinoma with morphological and immunohistochemical features overlapping those observed in 2002. The clinical situation worsened and the patient died in November 2009. The clinical aspects and the problems in the differential diagnosis are discussed.

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Section: Discussionmentioning

confidence: 91%

Cushing’s Syndrome due to a Pancreatic Neuroendocrine Tumor Metastatic to the Ovaries: a Clinicopathological Description of a Case

Rosa¹,

Marando

Ghezzi

et al. 2011

Endocr Pathol

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“…Sources of ectopic ACTH production arising in the pelvis are rare although EAS has been described due to gonadal tumours (adenocarcinoma, androblastoma, Sertoli cell tumour, carcinoid tumour and teratoma) ( 4 , 5 , 6 ) and genitourinary sources including the prostate ( 7 ) as well as cloacogenic carcinoma of the anal canal ( 8 ). Prostate cancer causing CS due to ectopic ACTH or CRH production is rare with <30 published cases.…”

Section: Introductionmentioning

confidence: 99%

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Elston

Crawford

Swarbrick

et al. 2017

Endocrine Connections

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Cushing’s syndrome (CS) due to ectopic adrenocorticotrophic hormone (ACTH) is associated with a variety of tumours most of which arise in the thorax or abdomen. Prostate carcinoma is a rare but important cause of rapidly progressive CS. To report a case of severe CS due to ACTH production from prostate neuroendocrine carcinoma and summarise previous published cases. A 71-year-old male presented with profound hypokalaemia, oedema and new onset hypertension. The patient reported two weeks of weight gain, muscle weakness, labile mood and insomnia. CS due to ectopic ACTH production was confirmed with failure to suppress cortisol levels following low- and high-dose dexamethasone suppression tests in the presence of a markedly elevated ACTH and a normal pituitary MRI. Computed tomography demonstrated an enlarged prostate with features of malignancy, confirmed by MRI. Subsequent prostatic biopsy confirmed neuroendocrine carcinoma of small cell type and conventional adenocarcinoma of the prostate. Adrenal steroidogenesis blockade was commenced using ketoconazole and metyrapone. Complete biochemical control of CS and evidence of disease regression on imaging occurred after four cycles of chemotherapy with carboplatin and etoposide. By the sixth cycle, the patient demonstrated radiological progression followed by recurrence of CS and died nine months after initial presentation. Prostate neuroendocrine carcinoma is a rare cause of CS that can be rapidly fatal, and early aggressive treatment of the CS is important. In CS where the cause of EAS is unable to be identified, a pelvic source should be considered and imaging of the pelvis carefully reviewed.

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“…In our literature search we have found only 2 other well-described cases caused by primary ovarian carcinoma (Crawford et al, 1994;Ball et al, 1996), though the condition had been mentioned previously (Jex et al, 1985). Ectopic ACTH syndrome has been reported in other types of ovarian tumour, including carcinoids (Brown and Lane, 1965;Gabrilove et al, 1967;Schlaghecke et aI., 1989), malignant tumour of the lipid (steroid) cell group (Marieb et al, 1983), Sertoli-cell carcinoma (Nichols et al, 1962), teratoma (Axiotis et al, 1987), and sex cord stromal tumours (Azzopardi and Williams, 1968;Kasperlik-Zaluska et al, 1993), respectively.…”

Section: Discussionmentioning

confidence: 97%

Ectopic ACTH-Syndrome due to ovarian carcinoma

Orbetzova¹,

Andreeva²,

Zacharieva³

et al. 2009

Exp Clin Endocrinol Diabetes

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The case of a 60-year-old female with severe and rapidly progressing Cushing's syndrome with fatal consequences is described. Both the clinical and the biochemical findings were consistent with ectopic ACTH production. A computed tomographic (CT) scan revealed intact pituitary and enlarged adrenal glands. Liver metastases were seen but the primary neoplasm was not found. Treatment with aminoglutethimide and other therapeutic measures were unsuccessful. A carcinoma of the right ovary was discovered on autopsy. The tumour cells showed immunoreactivity for neuron-specific enolase (NSE), ACTH and beta-endorphin with differing degree of intensity.

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Ectopic ACTH syndrome due to bilateral ovarian androblastoma with double, gynandroblastic differentiation in one ovary

Cited by 12 publications

References 13 publications

Cushing’s Syndrome due to a Pancreatic Neuroendocrine Tumor Metastatic to the Ovaries: a Clinicopathological Description of a Case

Cushing’s Syndrome due to a Pancreatic Neuroendocrine Tumor Metastatic to the Ovaries: a Clinicopathological Description of a Case

Severe Cushing’s syndrome due to small cell prostate carcinoma: a case and review of literature

Ectopic ACTH-Syndrome due to ovarian carcinoma

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