A 36-year-old man presented with recurrent urinary obstruction and an enlar
ged, partially cystic prostate tumour on ultrasonography. Microscopically,
the tumour was composed of cystically dilated ducts with leaf-like stromal
projections in the lumen. A portion of the stroma was very cellular with at
ypia and a high mitotic rate. In addition, there was a small focus with wel
l differentiated rhabdomyoblasts. This case is the second description of a
cystosarcoma phyllodes of the prostate with rhabdomyoblasts differentiation
. With radical surgery and chemotherapy only partial remission could be obt
ained.