Human dirofilariasis is a zoonotic infection most commonly caused by Dirofilaria repens. It has not been widely recognized in India. There is probably a focus of human infection with D. repens in Kerala. We report the first case of dirofilariasis, from the Eastern-part of India, to the best of our knowledge. Among the documented cases of human dirofilariasis caused by D. repens, recorded in India, most of them had ocular infections and few had subcutaneous involvement of the face. This is the first case report of human dirofilariasis from India involving the lower part of human body.