Human dirofilariasis is a rare infection caused by filarial worms of genus Dirofilaria. Humans may be infected as aberrant (accidental) hosts, mainly by Dirofilaria repens and Dirofilaria immitis. Most of the cases reported in India are of ocular dirofilariasis, with a few reports on subcutaneous dirofilariasis. We hereby report one such case of human subcutaneous dirofilariasis affecting the neck region of a 63-year male patient from Assam. This case indicates that this infection is a potentially emerging zoonosis in Assam and there is scope of finding more such cases with surveillance.