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Endocrine Abstracts (2018) 56 P52 | DOI: 10.1530/endoabs.56.P52

ECE2018 Poster Presentations: Adrenal and Neuroendocrine Tumours Adrenal cortex (to include Cushing's) (70 abstracts)

Bilateral adrenalectomy for occult ectopic Cushing’s syndrome in two patients with catastrophic hypercortisolism

Oya Topaloglu 1 , Nagihan Bestepe 1 , Bulent Yalcin 2 , Mehmet Kilic 3 , Melike Rusen Metin 4 , Gulin Ucmak 5 , Reyhan Ersoy 1 & Bekir Cakir 1


1Department of Endocrinology and Metabolism, Yildirim Beyazit University School of Medicine, Ankara, Turkey; 2Department of Medical Oncology, Yildirim Beyazit University School of Medicine, Ankara, Turkey; 3Department of General Surgery, Yildirim Beyazit University School of Medicine, Ankara, Turkey; 4Department of Radiology, Ataturk Education and Research Hospital, Ankara, Turkey; 5Department of Nuclear Medicine, Oncology Research and Training Hospital, Ankara, Turkey.


Introduction: Ectopic adrenocorticotropic hormone (ACTH) secretion is a less common cause of Cushing syndrome(CS) and is seen in 5 to 10% of patients with endogenous hypercortisolism. The most common types are bronchial carcinoids and small cell lung carcinoma. However, in approximately 10–20% of the cases, overt tumor cannot be found. Here, we described two patients with catastrophic hypercorticolism associated with ectopic CS and who were treated with bilateral adrenalectomy.

Case 1: A 47-year-old female patient had a history of a total abdominal hysterectomy and bilateral salpingo-oopherectomy operation due to clear cell ovary carcinoma 7 months ago. She was treated with chemotherapy. After 3rd dose chemotherapy, she had been hospitalized and treated due to sepsis associated with urinary tract infection. During the follow-up period in oncology, hypokalemia was detected and she was referred to our department due to severe muscle weakness in lower extremities and hypokalemia (serum K: 2.2 mEq/l). 24-h urinary free cortisol, ACTH measurement, dexamethasone suppression test (DST) demonstrated an ACTH dependent hypercortisolism. The inferior petrosal sinus sampling was indicative for an ectopic ACTH secretion. CT-scans of the thorax and abdomen, FDG-PET/CT scan and Ga68 DOTATATE PET/CT scans, were unable to demonstrate malignancy. Previous ovarian pathology was screened for ACTH secretion and evaluated as negative. Antifungal and metyrapone were started and then bilateral adrenalectomy was performed.

Case 2: A 71-year-old male patient who had a medical history of metastatic prostate carcinoma was referred to our department due to severe hypopotasemia, muscle weakness, refractory hypertension, peripheral edema. He had a history of second operation 3 months ago due to tumoral enlargement at the base of the urinary bladder and it was evaluated as infiltration of the prostate carcinoma. 24-h urinary free cortisol, ACTH measurement, DST demonstrated an ACTH dependent hypercortisolism. CT-scans of the thorax and abdomen, FDG-PET/CT scan and Ga68 DOTATATE PET/CT scans, were negative for malignancy. Medical therapy was started. But refractory hypertension, parenteral potasium infusion need were not suspended. Bilateral adrenalectomy was perfomed. He died due to pulmonary embolism 2 months after adrenalectomy.

Conclusion: Common treatmant options of Cushing syndrome consist of tumor management, somatostatin analogs, steroidogenesis inhibitors. Bilateral adrenalectomy is a highly effective treatment for patients with severe hypercortisolism if rapid control of hypercortisolism is desired. Mortality is high especially in patients with severe co-morbidities and mostly it depends on the prognosis of the underlying malignant tumor.

Volume 56

20th European Congress of Endocrinology

Barcelona, Spain
19 May 2018 - 22 May 2018

European Society of Endocrinology 

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