The Intriguing Case of a Double Pituitary Adenoma

When distinct pituitary hypersecretory manifestations coexist, the differential diagnosis includes plurihormonal or double pituitary adenomas. We describe a rare case of hypercortisolemia and hyperprolactinemia caused by 2 noncontiguous adenomas that required 2 surgeries.

A 37-year-old woman presented with 6 months of weight gain, amenorrhea, joint pain, leg swelling, and skin changes. She received prednisone for possible systemic lupus erythematosus. Four months later, she presented with headaches and new-onset diabetes with glucose >1000 mg/dL. Work-up revealed a right-sided 1.1-cm pituitary adenoma and prolactin level of 152.9 ng/mL (normal: 3-27 ng/mL). She was advised to stop the prednisone, start bromocriptine, and see a pituitary specialist. Examination revealed centripetal obesity, supraclavicular and dorsocervical fat pads, violaceous wide striae, bilateral leg edema, and galactorrhea. Workup confirmed adrenocorticotrophic hormone-dependent Cushing syndrome, with a central-to-peripheral gradient on inferior petrosal sinus sampling bilaterally. Transsphenoidal adenenomectomy yielded an adenoma diffusely positive for prolactin. Postoperatively prolactin normalized, hypercortisolemia persisted, and magnetic resonance imaging findings raised suspicion for a 2-mm microadenoma. The patient underwent a second operation when an adrenocorticotrophic hormone-positive adenoma was identified. After 4 years, both hypersecretory syndromes remain in biochemical remission.

A complete clinical and biochemical evaluation is necessary in patients with pituitary adenomas. Repeat surgery may be necessary for noncontiguous double adenomas.