Abstract Filarial infections are common in most tropical and subtropical regions of the world. We report a case of chyluria due to lymphourinary fistula in a filarial antigen negative case, the diagnosis of which was confirmed by the demonstration of microfilariae in urine as well as in peripheral blood only after diethyl carbamazine (DEC) provocation test. So In endemic areas, workup of filarial infection should be considered in a patient with chyluria even if the filarial antigen is non-reactive and microfilaria cannot be demonstrated initially.