Endocrine Abstracts

Introduction: We report a case of adrenal tumor recurrence after bilateral adenomectomy for Cushing’s syndrome.

Case report: A 44-year-old female diagnosed in June 2014 with Cushing’s syndrome caused by bilateral adrenocortical tumors, which were identified by an abdominal CT (right tumor-2.6/ 2/1.8 cm, left tumor-3/2/2 cm). At presentation, the patient had typical signs and symptoms of hypercortisolism and complications such as: arterial hypertension, dyslipidemia, severe osteoporosis with fragility rib fracture. Biological: an altered circadian cortisol rhythm (0800 h cortisol-19 μg/dl, 1100 h cortisol-28 μg/dl), decreased ACTH (ACTH<5 pg/ml) and unsuppressed cortisol by either low and high doses of dexamethasone (cortisol 24.3 μg/dl after 1 mg DXM and 16.6 μg/dl after DXM 2×2, respectively). As adrenal venous sampling showed that the right adrenal mass has secreted more cortisol than the left one, we decided to perform right adrenal adenomectomy (histopathological result showed nodular adrenal hyperplasia). The postoperative follow-up revealed persistent hypercortisolism and left adrenal adenomectomy was performed as well (anatomopathological examination confirmed adrenocortical adenoma). We initiated the replacement therapy with Hydrocortisone 25 mg/day for the functional adrenal insufficiency. The patient was monitored every 6 months, but there were no significant changes in clinical, hormonal or CT characteristics until the last re-evaluation (October 2017) when the CT scan performed showed a tumoral recurrence of 20/13 mm size in the extern arm of right residual adrenal gland and, also multiple hypoenhancing images dispersed in the sixth segment of the liver, as well as several pulmonary nodules in the lower part of both lungs. The hormonal investigations were unaffected, so the patient continued the glucocorticoid replacement therapy. These findings raised the need for extensive evaluation for digestive malignancy and the adrenal neoplasm could not be excluded as well.

Conclusion: The follow-up is mandatory in patients with nodular hyperplasia, as adrenal adenomectomy may not solve the problem and recurrences may occur even after several years.