Visceral leishmaniasis (VL) is a form of leishmaniasis, which causes significant mortality if untreated. The coexistence of VL with
Blastocystis
infection has not been well-documented in the literature. In this paper, we present the case of a 72-year-old male who experienced four months of recurrent diarrhea and later developed weight loss, fever, night sweats, and pancytopenia. The stool ova and parasite (O&P) examination revealed
Blastocystis
spp. vacuolar bodies and he was treated with metronidazole which resolved the diarrhea but not other symptoms. Further evaluation, including an abdominal Computed Tomogram (CT) scan and ultrasonography (USG), revealed splenomegaly. A splenic biopsy confirmed VL with numerous
Leishmania
amastigotes. Treatment with Amphotericin B led to clinical improvement. This paper discusses the clinical and diagnostic features of VL and
Blastocysti
s, highlighting their differential diagnosis, and available treatments.