ESPE Abstracts (2021) 94 P2-374

ESPE2021 ePoster Category 2 Pituitary, neuroendocrinology and puberty (48 abstracts)

Crooke Cell Adenoma as a Rare Cause of Recurrent Cushing Disease: A Challenge in Treatment and Follow-up

Arzu Jalilova 1 , Samim Ozen 1 , Cem Callı 2 , Erkin Ozgiray 3 , Yeşim Ertan 4 , Damla Gökşen 1 & Şükran Darcan 1


1Ege University Faculty of Medicine, Department of Pediatric Endocrinology, Izmir, Turkey; 2Department of Radiology, Ege University Faculty of Medicine, Izmir, Turkey; 3Department of Neurosurgery, Ege University Faculty of Medicine, Izmir, Turkey; 4Departments of Pathology, Ege University Faculty of Medicine, Izmir, Turkey


Introduction: Crooke’s cell adenoma (CCA) is an aggressive subtype of corticotroph adenoma, it is usually large, frequently invades surrounding tissues, and is resistant to both surgery and radiotherapy.

Case Report: A 13-year-old girl, admitted with weight gain for more than 3 months. Her height was 147.5 cm (-1.5 SDS), weight 58.5 kg (1.1 SDS), BMI 26.89 kg/m2 (2 SDS) and blood pressure was 100/78mmHg. Her i physical examination revealed truncal obesity, buffalo hump, moon-shaped face, acanthosis nigricans, abdominal striae and Tanner stage IV puberty. Early morning fasting blood glucose, lipids and thyroid function tests were normal, but serum cortisol was elevated and had lost its diurnal rhythmicity. 24-hour free urine cortisol level was high: 193 ug/l (4-56). The serum cortisol level was not suppressible (20.1 μg/dL) by low-dose dexamethasone, but was suppressed to 1.09 μg/dL by high-dose dexamethasone test. A pituitary magnetic resonans imaging (MRI) did not reveal a discrete adenoma and bilateral inferior petrosal sinus sampling showed that the pituitary was the source of Cushing syndrome. The interpetrosal sinus ACTH gradient indicated ACTH to the right side. Transsphenoidal right-sided pituitary adenomectomy was performed, and pathology result revealed Crooke cell adenoma. Postoperatively she had a normal pituitary reserve, with the exception of hypothyroidism and was commenced on thyroxine therapy. Three years later, she re-presented with rapid weight gain, menstrual cycle irregularity. In this time, her weight was 82.8 kg (3.0 SDS), height 156.6 cm (-1.09 SDS) and BMI was 33.7 kg/m2 (3.2 SDS). Early morning ACTH and cortisol levels were 51.8 pg/m L and 23.9 μg/dL respectively. Her 24-hour urinary free cortisol level was 46.26 ug/l (3-43). MRI examination revealed postoperative changes and 5x5 mm adenoma on the right hemisphere of the hypophysis. Cabergoline treatment was initiated for the patient. However, there was no response to the treatment within 3 months and the operation was planned for the second time.

Conclusion: Crooke’s cell adenoma is a rare cause of Cushing’s disease, which is difficult to treat and has a high risk of recurrence.

Volume 94

59th Annual ESPE (ESPE 2021 Online)

Online,
22 Sep 2021 - 26 Sep 2021

European Society for Paediatric Endocrinology 

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