In March 2006 a 64-year-old farmer complained of a 3-month-history of bilateral enlargement of the breasts, occasionally with blood in the urine. A brother had died in testis cancer at the age of 30. Blood samples from the present patient demonstrated very high plasma values of β-human chorion gonadotropin (β-hCG), estradiol and testosterone. He was on no drugs. The patient was referred to an endocrinologist. Physical examination demonstrated a moderate bilateral gynecomastia but normal testes. A CT of the chest and abdomen demonstrated no tumour-like lesions. However, an irregular thickening (5×1.6 cm) in the mid-line of the urinary bladder was found. An octreotide scintigraphy was normal. An ultrasound of the testes was normal. At cystoscopy a small exophytical broad-base tumour was observed in the posterior wall of the bladder but no satellite lesions. Biochemical assays still demonstrated high plasma values of total- and βhCG, estradiol and testosterone. Plasma gonadtropin values were found to be suppressed. An urinary steroid profile showed increased testosterone production.
In May a transurethral resection of the bladder tumour was performed. A histological examination demonstrated an urothelial cell carcinoma (WHO grade 3, without invasive growth) with foci of syncytial trophoblasts. These neoplastic parenchymal cells were found to be immunoreactive to βhCG. One week later plasma levels of total- and βhCG was marginally elevated; likewise estradiol was normalized and testosterone was low. The urinary steroid profile was also normal. Within in a month the gynecomastia completely disappeared. Another CT of the chest and abdomen showed just normal features.
In August plasma levels of total- and βhCG, estradiol and testosterone had returned to normal. Plasma gonadotropin values were only barely measurable.
Another cystoscopy with biopsies demonstrated no recurrence of the carcinoma. The patient felt healthy.
Although, admittedly, an ectopic production of hCG from a urinary bladder urothelial carcinoma is a rare cause of gynecomastia, it, nevertheless, can nowadays be considered as a fairly well established syndrome. The ability to differentiate in the direction of syncytial trophoblasts in an urothelial carcinoma might well have a genetical background. Be that as it may, the case history of the present patient shows that plasma assays of hCG is an excellent tumour marker in this proposed syndrome.
Tumour marker in this propsed syndrome.