Dirofilaria repens is a zoonotic mosquito-borne filarioid that parasitizes in the subcutaneous tissues of carnivores, that has been incriminated in the great majority of human dirofilariasis cases reported throughout the Old World. From the Republic of Uzbekistan, recent data showed that this parasite is endemic in domestic and wild carnivores. In contrast, information regarding human infection is scarce and mostly outdated, with a single case report being published during the last decades. The present paper reports a case of recurrent autochthonous infection with D. repens in a patient living in the rural area of the Sirdaryo Region of Uzbekistan and having no international travel history. The first diagnosis was in 2019, when the patient presented with what was suspected to be a tumour of the left breast. Three years later, in 2022, she presented for a consultation after experiencing redness, occasional swelling and persistent itching in the umbilical region of the abdomen. The ultrasound indicated the presence of a living, motile nematode. Following anthelmintic therapy, the parasite was surgically removed and identified as a female D. repens. The present report highlights the possibility of human re-infection with D. repens and further underlines the necessity of raising public awareness and implementing of solid control strategies in carnivores.