We present an unusual case of painful gynaecomastia due to a large adrenocortical tumour secreting oestradiol and other steroid hormones.
A 46 years old man presented with a 6 months history of progressive, painful gynaecomastia. He had no other specific symptoms and had previously been well. Alcohol intake was not excessive. Interestingly, his sister had presented with a phaeochromocytoma 2 years previously.
Examination demonstrated bilateral gynaecomastia, numerous spider naevi, and palmar erythema. The liver and spleen were not palpable and no jaundice or ascites were evident. Beard, axillary and pubic hair was present. External genitalia were unremarkable. There were no features of Cushings syndrome.
Serum concentrations of several steroid hormones were elevated: oestradiol at 887 pmol/l, testosterone 49.3 nmol/l, DHEAS 102.5 umol/l, and androstenedione >400 nmol/l. Urine free cortisol was 835 nmol/24 h, and serum cortisol at 0917 h was 580 nmol/l after overnight suppression with 1 mg of dexamethasone. Gonadotrophins were fully suppressed. Urine catecholamine excretion was normal. CXR showed a raised left hemidiaphragm, and CT scan confirmed the presence of a 17 cm left adrenal mass.
The left adrenal tumour was excised with perioperative corticosteroid cover. Serum hormone concentrations postoperatively included: oestradiol 95 pmol/l, testosterone 13.0 nmol/l, DHEAS 0.4 umol/l, LH 2.9 IU/l and FSH 3.1 IU/l. The patients gynaecomastia and other features of abnormal oestrogenisation resolved.
This case presents a number of points which merit consideration. These include:
A rare cause of rapid onset gynaecomastia.
Rapid growth of the tumour to a large size, as reported previously.
The possibility that the raised serum testosterone concentrations reported preoperatively might have resulted from an analytical interference in the presence of very high concentrations of DHEAS.
The unusual association with a family history of phaeochromocytoma.
03 - 07 May 2008
European Society of Endocrinology