We carried out a retrospective and descriptive study of 4 HIV infected patients with relapsing visceral leishmaniasis (VL) seen at 2 tertiary-care hospitals in Spain during the last 6 y, in whom miltefosine was used as a compassionate use treatment at a dosage of 50 mg b.i.d. Patients had a medium CD4 lymphocyte count of 69 cells/µl and were C stage. All patients received at least 2 different anti-leishmanial drugs and had at least 3 relapses before miltefosine treatment (range 3–7). Three patients were treated with miltefosine at a standard dose of 50 mg b.i.d. for 28 d, and the other during 12 months. Despite an initial symptomatic improvement, miltefosine treatment failed to eradicate the infection in all cases. We conclude that the use of miltefosine alone is not strong enough to cure relapsing VL in HIV-1 controlled infected patients.