A Rare Case of Dual Cortisol Sources: Pituitary and Adrenal Adenomas in Cushing’s Syndrome

Introduction This case reveals rare clinical case of autonomous cortisol production bilateral adrenal adenomas because of longstanding pituitary overstimulation, posing diagnostic and treatment challenges. Case Presentation D.B., a 64-year-old woman, was referred to an endocrinologist at the Hospital of Lithuanian University of Health Sciences Kaunas Clinics in February 2023 for treatment-resistant hypertension (HTN). She also had complaints of progressive weakness, fatigue, muscle pain, facial fullness, abdominal obesity, and HTN, prompting an evaluation for Cushing’s syndrome (CS). Abdomen CT scan revealed bilateral adrenal adenomas (BAAs). Elevated midnight saliva cortisol, 24-hour urine cortisol, and 1 mg, low dose and high dose dexamethasone suppression tests and higher ACTH finally confirmed ACTH-dependent Cushing’s disease (CD). MRI showed a pituitary microadenoma (ACTH-oma). In June 2023, she underwent transsphenoidal surgery, which confirmed ACTH-oma. Despite surgery, high cortisol levels persisted. ACTH levels were low. Repeated head MRI showed remission, abdomen MRI - stable BAAs The patient is under continued monitoring for recurrent hypercortisolism from adrenal - CS. Discussion CD accounts for 70% of endogenous CS cases [1]. Adrenal adenomas are rarer, and the coexistence of both, as seen here, is highly unusual [2]. Prolonged ACTH excess can lead to adrenal hyperplasia and, occasionally, cortisolproducing adenomas [3]. Persistent hypercortisolism with low ACTH post-surgery suggests adrenal autonomy. Further evaluation may include adrenal venous sampling (AVS) or genetic testing (ARMC5, MEN1) [4,5]. Persistent hypercortisolism and low ACTH post-surgery suggests adrenal involvement, which may require bilateral adrenalectomy or medical treatment [9–11]. Prognosis depends on cortisol control. Successful treatment reduces cardiovascular and metabolic risks, but persistent hypercortisolism increases morbidity and mortality. Bilateral adrenalectomy provides definitive control but requires lifelong hormone replacement [12]. Conclusions This case highlights the complexity of managing persistent hypercortisolaemia post-pituitary surgery. Further investigation into genetic causes and consideration of adrenostatic treatments are essential to improve the patient's outcome.