Patients with Cushing's disease have high levels of circulating corticosteroids. Treatment may result in unmasking of steroid dependant conditions. We report a case of ulcerative colitis presenting after bilateral adrenalectomy.
A 19 year old girl underwent bilateral adrenalectomy in November 1998 for pituitary dependant Cushing's disease. She was discharged on hydrocortisone and fludrocortisone and post operatively her cortisol day profile was satisfactory. Pre operatively she had no abdominal symptoms. 3 years later she presented with diarrhoea and abdominal discomfort. 24 hour urinary 5 HIAA and catecholamines were normal. Rectal biopsy was consistent with ulcerative proctitis and she was started on Asacol and Iron tablets. However in February she was admitted with bright red per rectal bleeding, tachycardia and hypotension. She needed 6 units blood transfusion and was started on prednisolone. As her symptoms deteriorated despite being on prednisolone 40 mg and Asacol, Azathioprine(steroid sparing agent) was added to her regime. Despite maximal medical treatment her colitis deteriorated and in October 2003 she needed total colectomy. Post operatively she improved, prednisolone dose was tapered off and is presently stable on hydrocortisone and fludrocortisone.
To our knowledge, this is the first report of ulcerative colitis being detected in a patient after bilateral adrenalectomy for pituitary dependant Cushing's disease. It is interesting to note that when her Cushing's disease was cured, her endogenous steroid levels fell, which is when the ulcerative colitis flared up. Corticosteroids are effective in both ulcerative colitis and Crohn's because of their anti-inflammatory and immunosuppressive properties.
We suggest that this case should raise awareness of the possibility of unmasking steroid dependant conditions after treatment of Cushing's disease/syndrome. Review of literature suggests that such cases are rare. Cases of sarcoidosis, Thyroiditis and inflammatory polyarthritis presenting shortly after surgical treatment of Cushing's syndrome has been reported
04 - 06 Apr 2005
British Endocrine Societies