Human dirofilariasis is a parasitic disease, transmissible helminthiasis caused by parasitizing nematodes of the genus Dirofilaria, most often D. repens and D. immitis which occurs with a predominant lesion of the skin (63%), conjunctiva (50%), mucous membranes, subcutaneous tissue and less often – internal organs. The most frequent incidence of dirofilariasis is observed in regions with a warm, humid climate, and at temperatures below 14℃, dirofilariae stop developing. Human cutaneous dirofilariasis on the European continent is most often diagnosed in representatives of southern and eastern Europe. There are confirmed cases of dirofilariasis on the territory of the Republic of Belarus and neighboring countries. Quite often, this disease affects the maxillofacial region. The purpose of the work was to initiate infectious alertness when planning operations for the removal of benign neoplasms in the maxillofacial region and inform medical specialists about the possibility of developing dirofilariasis of the specified localization, its clinical manifestations and objective diagnostic methods. Material and methods. The object of observation was a 34-year-old female patient R. with maxillofacial dirofilariasis. The subject of the study is the medical record of an inpatient patient R. Results and discussion. At the pre-hospital stage, patient R. underwent a general clinical examination. All indicators of the tests were within the age norm. According to the results of radiation examination methods, no data confirming the parasitic etiology of the disease was obtained. Taking into account the presence of a rounded formation about 1.0-1.2 cm in diameter which is slightly displaced relative to the underlying and surrounding soft tissues, the patient was offered surgical treatment. During the operation, a rounded self-moving helminth was extracted from the formation, about 11.0 cm long and 0.15 cm in diameter. After the helminth was identified, the patient was diagnosed with dirofilariasis. This should be taken into account in the diagnostic and therapeutic aspects when working with patients who have tumor processes in the maxillofacial region. Conclusion. The presented clinical observation shows that in the practice of a dental surgeon and a maxillofacial surgeon, it is increasingly possible to deal with the localization of helminth in the maxillofacial region which simulates a neoplasm which must be taken into account when conducting diagnostics, differential diagnosis and preoperative preparation. The results of the described case are aimed at developing infectious alertness when planning surgical interventions to remove benign neoplasms in the maxillofacial region, developing a rational diagnostic scheme and differential diagnosis for patients with suspected dirofilariasis and other parasitic lesions of the maxillofacial region