Leishmaniasis is a rare disease in both solid-organ and hematopoietic stem cell transplantation. Additionally, the frequency of disease is likely related to the leishmaniasis prevalence in the general population. Although cutaneous leishmaniasis (CL) presentation is more prevalent than that of visceral leishmaniasis (VL) in the general population, the opposite occurs in transplant patients. The current available knowledge is based on small series, case reports, or extrapolations from studies conducted in the general population or in immunocompromised subjects. Therefore, recommendations for disease management are based on the opinion of experts. Leishmania transmission through organ grafts has not been conclusively demonstrated, and there are relatively low rates of transfusion-transmitted leishmaniasis (TTL). To date, compulsory screening for VL in organ donors and the deferral criteria have still not been established. In the context of transplant recipients, diagnosis and management remain a challenge. Delayed diagnosis frequently occurs because of low clinical suspicion and atypical disease presentation. Liposomal amphotericin is the drug of choice and the most commonly used therapy. However, patient profile, Leishmania species, disease extent, drug availability or toxicity, presence of concomitant infections, and especially response can influence this choice. Immunosuppression dose reduction is often recommended, but such decisions must be taken on an individual basis. The main challenges include donor screening and organ deferral, duration of therapy, secondary prophylaxis, and follow-up.