MON-345 Recovery of Adrenal Function Following Bilateral Adrenal Hemorrhage: Two Case Reports

Background: Spontaneous or traumatic bilateral adrenal hemorrhage is a rare but potentially life-threatening condition which usually results in permanent adrenal insufficiency requiring life-long glucocorticoid and mineralocorticoid replacement. We present two patients with bilateral adrenal hemorrhage who subsequently partially recovered adrenal function. Case 1: A 38-year-old male developed bilateral adrenal hemorrhage and insufficiency after a car accident. His morning cortisol at the time of diagnosis was 0.6 mcg/dl (reference range 4.3-22.4 for morning cortisol) and his ACTH was 595 pg/ml (normal 7-69). He was treated with hydrocortisone and fludrocortisone. Hydrocortisone was discontinued 6 months later when he was found to have a morning cortisol of 10.8 mcg/dl. Fludrocortisone was discontinued a few months later when he developed hypokalemia and hypertension. His ACTH initially remained elevated at >500 pg/ml; and he did not experience a rise in his cortisol concentration after stimulation with 250 mcg of Cosyntropin, suggesting maximal baseline adrenal stimulation. He remained off glucocorticoid and mineralocorticoid replacement but was prescribed stress glucocorticoid coverage. Over the subsequent five years, his ACTH progressively returned to the normal range with recent level of 60 pg/ml with a morning cortisol of 13.6 mcg/dl. Case 2: A 67-year-old male developed bilateral adrenal hemorrhage secondary to heparin induced thrombocytopenia and presented with acute primary adrenal insufficiency (morning cortisol and ACTH at time of diagnosis were 5.5 mcg/dl and 219 pg/ml respectively). He was treated with hydrocortisone and fludrocortisone for 8 months. As he reported feeling well even on days when he missed his medications, his adrenal function was retested and he had a morning cortisol of 7.5 mcg/dl, and an ACTH of 452 pg/ml, suggesting partial recovery of adrenal function. Since his morning cortisol is still below 10 mcg/dl, he remains on glucocorticoid and mineralocorticoid replacement at lower doses, but we plan periodic re-evaluation of his adrenal function. Discussion: Adrenal dysfunction secondary to adrenal hemorrhage is generally believed to be irreversible, necessitating life-long glucocorticoid and mineralocorticoid replacement, except for a few reports in the literature suggesting partial recovery in some patients. We present two such rare cases in whom partial recovery of adrenal function was demonstrated after bilateral adrenal hemorrhage. Both patients had an unequivocal initial diagnosis based upon laboratory and imaging studies. Hence periodic re-evaluation of adrenal function should be considered in patients with adrenal insufficiency secondary to adrenal hemorrhage.