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Costa, Helce R. Julio-Junior, Fabio Bernardo, Francisco J. B. Sampaio, Luciano A. Favorito Background: The objective of this study is to analyze the incidence of congenital urogenital malformations in human fetuses with neural tube defects (NTDs). Methods: We studied 320 human fetuses (214 males and 106 female) ranged in age from 12 to 35 weeks post-conception (WPC). The fetuses were carefully dissected with the aid of a stereoscopic lens with 16/25× magnification. The abdomen and pelvis were opened to identify and expose the urogenital organs. We studied the incidence of renal, ureteral, bladder, urethral, testicular, epididymal, vas deferens, prostate and penile and uterine anomalies. The renal anomalies were divided into rotation, fusion, number and ascension. We observed the presence of seminalvesicle and ureteral duplication, bladderseptation, hypospadias and posterior urethral valve. Results: We observed 17 fetuses (5.31%) with NTDs ranged in age between 18 to 34 WPC, weighted between 170 and 2325 g, and had crown-rump length between 13 and 34 cm. Of the 17 fetuses, 4 (23.4%) presented some kind of anomaly of the urogenital system. Renal anomalies were found in three fetuses (17.6%) and a duplication of the left ureter in one fetus (5.8%). We do not observe anomalies in genital organs in this sample. Conclusions: We concluded that the urogenital anomalies in human fetuses with neural tube defects are significant with an incidence superior a 20%. In this study we have not found severe urogenital anomaly that causes damage of the body function. 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