Zoonotic filariasis caused by Dirofilaria repens(D. repens) is prevalent in several regions of the world including Europe, Africa, and Asia. It is considered an emerging zoonosis, due to the recent rise of human D. repens infections. In India, most of the documented cases of human dirofilariasis were from ocular infections, few from subcutaneous infections, but it was never reported from muscular plane. To the best of our knowledge, globally this is the first case of human D. repens infection caused by gravid worm in the muscle tissue.