We detected a high ZIKV RNA load and replicative ZIKV in semen samples, but ZIKV remained undetectable by rRT-PCR in the blood sample collected at the same time. These results suggest that viral replication may have occurred in the genital tract, but we do not know when this replication started and how long it lasted. The fact that the patient had no common symptoms of ZIKV acute infection concomitantly to hematospermia suggests that the viremic phase occurred upstream, probably during the first or second episode of mild fever, headache, and arthralgia.