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ABSTRACT: Objective: To report a case of a giant retroperitoneal teratoma involving the adrenal gland associated with persistent postoperative adrenal insufficiency likely due to the secretion of corticotropin releasing hormone (CRH) and adrenocorticotropic hormone (ACTH) from the teratoma.Methods: We describe the presenting signs and symptoms, investigations, and pathologic features leading to the diagnosis and treatment options.Results: A 29-year-old male was found to have a large retroperitoneal mass arising near the right kidney and adrenal, encasing the dual renal arteries and causing right urinary tract obstruction on computed tomography imaging. Resection of the mass required right nephrectomy and median sternotomy. Microscopy demonstrated a mature teratoma arising from the retroperitoneum. The right adrenal gland was compressed and trapped in the edge of the mass. His postoperative course was complicated by unexpected persistent hypocortisolism requiring cortisol replacement. Subsequent immunostaining was positive for CRH and ACTH, suggesting that the teratoma was secreting CRH and ACTH.Conclusion: This is the first reported case of teratoma associated with CRH and ACTH secretion leading to postoperative adrenal insufficiency. Although extremely rare, the possibility of a teratoma secreting hormones should be kept in mind and appropriate evaluation is recommended pre- and postoperatively.ACTH = adrenocorticotropic hormone; AFP = alphafetoprotein; CRH = corticotropin releasing hormone; CT = computed tomography; RR = reference range