Endocrine Abstracts

Introduction: Bilateral macronodular adrenal hyperplasia is a rare cause of Cushing’s syndrome (CS) characterized by multiple adrenal nodules >1 cm. It can be diagnosed in patients with overt CS but is more often diagnosed incidentally, especially in the 5th or 6th decades of life.

Clinical case: 61-year-old woman with type 2 diabetes, arterial hypertension and dyslipidemia treated with metformin+sitagliptin 1000/50 mg bid, valsartan+hydrochlorothiazide 160/25 mg qd, bisoprolol 2.5 mg qd, atorvastatin 20 mg qd. Irrelevant family history. After right hemicolectomy for a colon polyp with high-grade dysplasia she performed a CT scan which showed nodular thickening of the adrenal glands with bilateral hypodense nodules, the largest in the right adrenal gland with 32 mm and in the left with 31 mm. She was referred to the Endocrinology appointment. She complained of anxiety and fatigue. At physical examination: weight 66.8 kg, height 150 cm, BMI 29.7 kg/m², arterial pressure 159/93 mmHg, heart rate 77 bpm, central obesity, thin limbs; no reddish purple striae, facial plethora, easy bruising, dorsocervical fat pad or proximal myopathy. Biochemical study showed high serum cortisol levels after 1-mg overnight dexamethasone suppression test (DST) in two separate measurements (24.7 and 20.4 μg/dl), absence of cortisol suppression after the longer dose DST (serum cortisol 14.4 μg/dl), late-night serum cortisol of 11.4 μg/dl, normal urinary free cortisol in two separate measurements ((59.4; 34.5; 62.1 μg/day (N:4.3–176.0)) and morning serum ACTH of 1.0 ng/l in two measurements. Bone mineral density was normal. A follow-up CT scan showed enlargement of the adrenal glands, which contained multiple spontaneously hypodense and bilateral nodules, measuring the largest one 3 cm in the right side and 3.1 cm in the left. The nodules were well delimited, without interstitial calcifications, suggestive of multiple adrenal adenomas. The investigation for ectopic adrenal receptors was negative.

Discussion: This is a case of bilateral macronodular adrenal hyperplasia with autonomous cortisol secretion. The patient symptoms and comorbidities may be related to excessive cortisol secretion. In the absence of a clear picture of CS adrenalectomy is not recommended. However, unilateral adrenalectomy of the largest lesion is a possibility. In this case, since there was no overt CS, the comorbidities were controlled under medical treatment and there was similar involvement of both glands, we decided for active surveillance to avoid the consequences of bilateral adrenalectomy.