Background: Onchocerciasis is the world's second commonest infectious cause of blindness. It is transmitted by the bite of the Simulium blackfly, which transmits the infective-stage larva into the human skin and mainly affects the people in the rural areas of Sub-Saharan Africa, Yemen, and those living in parts of Central and South Africa. The first case of ocular onchocerciasis in India was a woman from rural Assam, northeastern India reported in 2011. Here we report a rare case of onchocerciasis—a patient from a non-endemic region of Bengaluru, India, showing a swelling in the orbital region. Case description: A 55-year-old women was presented to our outpatient department with the complaints of a swelling below the left eyebrow since 2 months and drooping of the left upper eyelid since 1 month. The patient underwent excision of the swelling after CT and FNAC of the swelling. The worm in the wall of the excised cyst was identified as Onchocerca volvulus, on the basis of morphological features observed in histopathology. The patient was treated with a single oral dose of Ivermectin (150 μg/kg), and on follow-up at 4 months, she has not shown any recurrence of symptoms or fresh complaints. Conclusion: Onchocerciasis, though rare, can be a differential diagnosis of a subcutaneous swelling in the body. Clinical significance: Knowledge and awareness regarding the presence of this filarial worm in India is required, as it is rare and reporting of further cases needs to be done.