We report the case of a 56-year-old patient, presenting with bilateral hypertensive anterior uveitis due to Leishmania infantum, in the setting of immune deficiency related to HIV infection. The etiology of the uveitis was diagnosed following detection of L. infantum DNA on anterior chamber paracentesis. Initially, the patient received an intravitreal injection of liposomal amphotericin B, systemic antiparasitic treatment, topical steroid and ocular hypotensive treatment. Due to unfavourable disease progression in the right eye, we re-evaluated the anti-inflammatory, antiretroviral and antiparasitic medications (beginning meglumine antimoniate): the uveitis in the left eye was thus able to be controlled. We followed the efficacy of treatment with weekly quantification of Leishmania DNA in the aqueous humor. Uveitis secondary to leishmaniasis is rare and serious. Physicians must be aware of this cause of uveitis, particularly in immunodeficient patients. The quantitation of Leishmania DNA in the aqueous humor is an indispensible tool for monitoring the disease.