Endocrine Abstracts

A 68-year-old man with history of non-muscle-infiltrating bladder tumor and atrial fibrillation was referred to the Endocrinology Department in November 2020 for a functional study of a left adrenal incidentaloma. Furthermore, the patient had a history of arterial hypertension diagnosed at the age of 45 years, on treatment with 3 drugs (enalapril 20 mg B.I.D., amlodipine 5 mg B.I.D., hydrochlorothiazide 12.5 mg/day), achieving usual blood pressure (BP) levels of 150/90 mmHg. At the first evaluation (November 2020) in the Endocrinology Department, headaches, frequent palpitations and non-weight gain in recent years were reported, BMI of 26 kg/m2 and no specific clinical data of hypercortisolism. Functionality study was requested, after replacing antihypertensive medication for at least 4 weeks with doxazosin 4 mg, obtaining the following results: plasma ACTH <3.8 pg/mL (normal range 7.7-48.8); serum basal cortisol 18.7 mg/dl (normal range: 3.7-19.4); urinary free cortisol (UFC) 75.00 mg/24 h (normal <140); Late-night salivary cortisol 4.62 nmol/l (<7,56); Cortisol after suppression with 1 mg of Dexamethasone 7.10 mg/dl. Aldosterone/renin ratio and metanephrines were normal. HbA1c was 6.1%. In September 2020, non-enhanced CT scan has showed radiological stability of the adrenal lesion with respect to 2018 imaging tests: 41x37mm, oval, sharp-edged, heterogeneous, with majority component of low-density value, compatible with lipid-rich adenoma. The study was extended to norcholesterol scintigraphy, and the findings were consistent with hyperfunctioning left adrenal adenoma. The patient refused adrenalectomy, opting for conservative management. During follow-up, home BP values were around 150/90 mmHg. Glycemic control worsened, HbA1c increasing over 6.5%. Treatment with metformin/empagliflozin was initiated. No osteopenia/osteoporosis was detected. Given patient’s refusal of surgery and coexistence of two comorbidities potentially associated with hypercortisolism, one of them poorly controlled, metyrapone treatment (currently off-label) following the Debono protocol was proposed. Upon approval by the hospital pharmacy, metyrapone was started at a night dose of 250 mg. After 2 weeks, BP improved notably, reaching 120/70 mmHg, and amlodipine was suspended. Metyrapone night dose was increased to 500 mg and BP continued falling, so enalapril was discontinued. After 3 months on treatment, pathology tests showed basal cortisol at 15.7 mg/dl; UFC at 31.20 mg/24 h; Late-night salivary cortisol at 3.18 nmol/l ; HbA1c at 6.6%. No secondary side effects were developed. We can conclude that metyrapone is a useful and safe treatment for patients with MACS and associated comorbidities, offering a better control of hypertension.