https://imcjms.com Ibrahim Medical College Journal of Medical Science https://imcjms.com/registration/journal_full_text/55 Tue, 02 Aug 2016 11:14:14 +0000 We present a case of Cushing’s syndrome and virilization in a 15 year old girl which was suspected to be due to an adrenal carcinoma. She presented with features of virilization in addition to those of hypercortisilism. Her high androgen levels especially dehydroepiandrosterone sulfate (DHEAS) were also in favor of an adrenal carcinoma. An unenhanced computerized tomography (CT) scan showed a mass (size: 5.3 cm) in the right adrenal gland with a soft tissue intensity of more than 10 HU which was suggestive of adrenal carcinoma. But, histopathology of the resected mass revealed a benign adrenocortical adenoma. Address for Correspondence: Dr. Tahniyah Haq, Department of Endocrinology, BIRDEM, 122 Kazi Nazrul Islam Avenue, Dhaka 1000, Bangladesh, E mail: tahniyahhaq@yahoo.com   A few cases of adrenal adenoma have been reported with features of virilization.1,2 As far as we know, previously no cases of virilizing adrenal adenoma have been reported in Bangladesh. We describe here a case of adrenal adenoma in a young Bangladeshi girl with features of virilization and a high androgen level. Case presentation On physical examination, she was noticed to have a round plethoric face with moderate acne and hirsutism. The Ferriman-Gallway score for hirsutism was 1 for upper lip, 4 for chin, 4 for trunk and limbs. There were purple striae over her axillae, abdomen and thigh (Figure 1a). Acanthosis nigricans was present over the axillae. Tanner stage was V for breast and IV for pubic hair. She had clitoromegaly (Figure 1b). No lump was palpable on abdominal examination, but there was tenderness over the right lumbar area. Her blood sugar fasting and after 75g of glucose meal was 5.7mmole/L and 10.5mmole/L respectively. Serum sodium level was 146mmole/L and S. potassium was 3.6mmole/L. All other biochemical and haematological parameters were unremarkable. Chest X-ray and ECG were normal. On March, 2011 endocrine evaluation showed a luteinizing hormone level of 0.64 mIU/ml (normal: 1.1-11.6 mIU/ml), FSH was 0.36mIU/L (2.8-11.3 mIU/ml). Her androgen levels were extremely elevated. Testosterone was 17.05nmol/L (0.89-4.22), dehydroepiandrosterone sulfate (DHEAS) was 458 µgm/dL (35-430). ). After 4 months, testosterone level rose even higher to 43.90nmol/L. 17-OH progesterone level was normal (3.92ng/ml). Serum cortisol at 9am was 1303.80nmole/L (116-1065) and at 5pm 1656nmole/L. Twenty four hour urinary cortisol was high (1937.52 nmole/L). Overnight dexamethasone suppression test was done. Next morning cortisol was 1139.84nmole/L. ACTH level was 33.8pg/ml (8.3-57.8). Patient was euthyroid as the FT4 and TSH levels were 10.94pmole/L and 0.91IU/ml respectively. An ultra sonogram (USG) of the whole abdomen did not reveal any abnormality. A repeat USG of abdomen 6 months later detected a hypoechoic mass measuring 5cm × 3.9cm in the right adrenal gland. Left adrenal gland and ovary were normal in appearance. The mass was evaluated further with a computerized tomography (CT) imaging. CT findings showed an oval shaped well circumscribed soft tissue density (HU 46) mass lesion measuring about 5.3cm × 4.7cm in the right suprarenal area (Figure 1c). After intravenous contrast there was minimum enhancement (HU 51). Flat plane around mass was well preserved. No perilesional structural involvement was seen. Left adrenal gland was normal.   Fig.1a: Wide, purple striae over lower abdomen, 1b: Clitoromegaly, 1c: Contrast CT showing an oval shaped well circumscribed soft tissue density (HU 51) mass measuring about 5.3cm × 4.7cm in the right suprarenal area with minimum contrast enhancement. 1d: H&E staining of resected tumor showing polygonal cells having round to oval nuclei and abundant eosinophilic cytoplasm. The case was diagnosed as adrenal carcinoma based on high androgen levels especially DHEAS and the presence of a mass (5.3 × 4.7cm) in the right adrenal gland having a soft tissue density of 46HU. Following surgery, menstruation started on the first postoperative day. Blood pressure came down to normal and anti-hypertensives drugs were stopped from the first postoperative day. The cushingoid features and hirtutism resolved over 6 months following surgery. The patient was maintained on steroids which was planned to be gradually withdrawn over months. Discussion Our patient presented with features of virilization such as hirsutism and clitoromegaly. She had markedly increased levels of DHEAS and unenhanced CT scan showed a 5.3 cm mass with an intensity of 46 HU (i.e. more than 10 HU). All these features suggested an adrenal carcinoma. However, histopathology later proved the tumor to be of benign nature. Therefore, the final diagnosis was adrenocortical adenoma.   1.   Dahms T W, Gray G, Vrana M, New M I.  A case presenting as Cushing syndrome with virilization. Am J Dis Child 1973; 125: 608-611. 2026 Ibrahim Medical College. All rights reserved.