A 70-year-old woman who developed a moon face was hospitalized. At admission, adrenocorticotropic hormone (ACTH) and serum cortisol levels were elevated. A high-dose dexamethasone suppression test demonstrated that cortisol secretion was uninhibited. Furthermore, chest computed tomography revealed an anterior mediastinal mass. On the basis of these results, ectopic ACTH syndrome was diagnosed and complete resection of the thymic tumor was performed. Histological examination revealed large cell neuroendocrine carcinoma of the thymus and positive immunoreactivity for ACTH. However, despite complete excision of the thymic tumor, ACTH and serum cortisol levels were high. Therefore, we investigated whether the lesion had recurred or metastasized; however, the results were inconclusive. Finally, laparoscopic bilateral adrenalectomy was performed to control the hypercortisolemia. After the surgery, the serum cortisol level was well-controlled and the general condition of the patient was good.
Background. Bilateral adrenal hemorrhage is a serious condition that can result in adrenal insufficiency, shock, acute adrenal crisis, and mortality if it is not managed with adequate treatment. We report a rare case of idiopathic bilateral adrenal hemorrhage.Case presentation. A 50-year-old woman visited our hospital with complaints of right upper abdominal pain. A computed tomography (CT) revealed unilateral left adrenal gland hemorrhage. However, the results of rapid adrenocorticotropic hormone (ACTH) stimulation test and adrenomedullary hormone function test were normal. Since the patient did not show signs of adrenal insufficiency, corticosteroid therapy was postponed and only supportive management therapy was started. After 1 week, a followup CT showed a previously unseen adrenal hemorrhage on the right adrenal gland, but the rapid ACTH stimulation test result was normal. One year later, no hemorrhagic signs were observed on the follow-up CT. Conclusion.In most cases of idiopathic bilateral adrenal hemorrhage, patients are treated with steroid replacement therapy due to adrenal insufficiency. In some other cases, patients are treated with steroids despite the absence of adrenal insufficiency. Here we reported a very rare case of idiopathic bilateral adrenal hemorrhage sequentially to emphasize that before initiation of adrenal hormone replacement therapy, it is important to determine whether adrenal insufficiency is present. If there is no evidence of adrenal insufficiency, adrenal replacement therapy should be postponed until the presence of adrenal insufficiency is confirmed.
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