Background: Recent studies have demonstrated an association between congenital Zika virus (ZIKV) infection and multiple birth defects, particularly microcephaly; however, to date, there have been no reports on the consequences of ZIKV infection on the development of the urinary tract. Methods & Materials: Herein, we reported on the first case of a pregnancy having ZIKV-related urinary bladder agenesis. Results: A 22-year-old patient who conceived her third pregnancy and became infected with Zika virus at 16 weeks' gestation in Sucre, Colombia. Zika infection was confirmed by real-time PCR; ultrasound revealed biometry with decreased fetal movements, and amniotic fluid decreased (index < 3) (severe oligohydramnios) and anterior placenta (I/III). In a new ultrasound at 17 weeks, an estimated weight of 168 gr was calculated, with anhydramnios and intrauterine growth restriction (IURG). At this point a complete absence of the urinary bladder was evidenced. Because of the poor prognosis, pregnancy was interrupted. The fetal and placental tissue samples for RT-PCR were positive for ZIKV. Conclusion: Given the originally not adverted wide teratogenic potential of ZIKV, the severe damage caused at urinary tract would be related to this flavivirus infection. Complete agenesis of urinary bladder is an extremely rare anomaly with only a few live cases reported so far. To understand the impact and mechanisms of ZIKV infection on human tissues development and the link of ZIKV to birth defects, a key step is to identify cell types that are particularly vulnerable to viral infection in the developing tissues after ZIKV breaches the placental barrier. This, particularly outside the central nervous system is not properly understood yet. Arthrogryposis, IURG, uveitis and retinal degeneration have been reported in association with gestational ZIKV infection. ZIKV epidemics has reminded us the fragility of the human beings to emerging infectious diseases, as previously experienced with many other agents. Moreover, ZIKV also changed the way researchers and physicians deal with flavivirus infections. This is due mainly to the severe impact of ZIKV infection during pregnancy and the resulting congenital ZIKV syndrome (CZS) and other birth defects that would include potentially the urinary tract abnormalities, as seen in our case.