Purpose To compare the structure of the testis in fetuses with prune belly syndrome (PBS) to normal controls. Materials and Methods We studied 6 testes obtained from 3 fetuses with PBS and 14 testes from 7 male fetuses. The testicular specimens were cut into 5-μm thick sections and stained with hematoxylin and eosin (HE), to observe the seminiferous tubules; Weigert's solution to observe elastic fibers; and picrosirius red to observe collagen. The images were captured with an Olympus BX51 microscope and Olympus DP70 camera. The stereological analysis was done with the Image Pro and Image J programs. Means were statistically compared using the Mann–Whitney U test (p < 0.005). Results Quantitative analysis documented no differences (p = 0.4) in number of seminiferous tubules (ST) in PBS testes (mean = 8.87%, SD = 1.59), when compared to the control (mean = 11.4%, SD = 2.99) and no differences (p = 0.8) in diameter of ST in PBS testes (mean = 52.85 μm, SD = 1.58) when compared to the control group (mean = 53.17 μm, SD = 1.55), but we did observe a lower number (p = 0.0002) of Leydig cells in the PBS testes (mean = 67.03% and SD = 3.697) when compared to the control group (mean = 90.1% and SD = 2.986). Conclusions Our study showed a lower concentration of Leydig cells in the triad syndrome fetuses.