Cushing syndrome with female virilization unraveling functional adrenocortical oncocytoma in a 15-year-old girl: A case report and review of current literature

Abstract

Introduction: Adrenal oncocytic tumors are rare, especially among children. They are often nonfunctioning and benign but can express with Cushing syndrome, female virilization, precocious pseudo-puberty, or hirsutism when they are hormonally active. However, complex association of amenorrhea with Cushing syndrome and hirsutism in female children is unusual. Case presentation: We present the case of a 15-year-old girl with recent amenorrhea, Cushing syndrome, and hirsutism. Blood tests showed elevated cortisol and androgens levels. Imaging studies showed a large mass in the left adrenal gland. After laparoscopic adrenalectomy, the histopathological examination diagnosed an adrenocortical oncocytoma categorized as benign by the Lin-Weiss-Bisceglia classification. The patient was discharged from hospital on the 4th postoperative day. After 16 months, she remains recurrence-free clinically and on imagery. Conclusion: To our knowledge, this clinical endocrine picture due to unusual co-secretion of cortisol and androgens by an adrenal oncocytic tumor is exceptional, particularly in children. Only three similar cases have been reported in the pediatric literature yet. However, this condition is amenable to surgery with complete resolution of abnormal clinical manifestations.

Keywords

• Adrenocortical
• Oncocytoma
• Cushing
• Hirsutism
• Case report