A patient with a diagnosis of chronic and idiopathic unilateral dacryoadenitis was submitted to our clinic for diagnostic reasons from a regional hospital. Within a period of 1 year, this was the second onset of a swelling in the region of the lacrimal gland which – unlike the first time – showed resistance to antibiotic treatment. The patient’s general and ophthalmic condition was good, and his only complaint was of cosmetic nature because of the disfiguring swelling in the lateral part of the left upper eyelid. Basic laboratory findings showed no abnormalities and radiographic imaging showed enlargement of the left lacrimal gland without bony destruction. Guided by epidemiological data for noninfectious dacryoadenitis we decided to perform excisional biopsy of the left lacrimal gland. To our surprise, the pathologist verified the presence of a worm of the genus <i>Dirofilaria,</i> which is, to the best of our knowledge, the second reported case of lacrimal gland dirofilariasis in the literature and the second reported case of such a disease in Croatia.