Gross haematuria caused by lymphatic filariasis has been rarely reported. An adult woman living in a filarial-endemic area presented at a hospital in Orissa, India, in July 2004, with painless gross haematuria without any associated symptoms, such as dysuria, abdominal pain and fever. Urine microscopy revealed many erythrocytes and the immunochromatographic test was positive for filarial antigenaemia. After excluding other causes of haematuria, the patient was treated with a standard dose of diethylcarbamazine for 12 days and a single dose of ivermectin (200 microg/kg) and responded well without any recurrence for 2 years of follow-up.