Tehran University of Medical Sciences Iranian Journal of Parasitology 1735-7020 16 1 2021 02 17 159 163 Mohammad Reza ABDOLSALEHI 1. Department of Infectious Diseases, Pediatrics Center of Excellence, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran Mehdi MOHEBALI 2. Department of Parasitology and Mycology, School of Public Health, Tehran University of Medical Sciences, Tehran, Iran 3. Center for Research of Endemic Parasites of Iran (CREPI), Tehran University of Medical Sciences, Tehran, Iran Hossein KESHAVARZ 2. Department of Parasitology and Mycology, School of Public Health, Tehran University of Medical Sciences, Tehran, Iran 3. Center for Research of Endemic Parasites of Iran (CREPI), Tehran University of Medical Sciences, Tehran, Iran Shima MAHMOUDI 4. Pediatric Infectious Disease Research Center, Tehran University of Medical Sciences, Tehran, Iran Setareh MAMISHI 1. Department of Infectious Diseases, Pediatrics Center of Excellence, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran . 4. Pediatric Infectious Disease Research Center, Tehran University of Medical Sciences, Tehran, Iran 2021 01 05 2021 02 17 Chronic granulomatous disease (CGD) described as an essential immunodeficiency problem of phagocytic cells resulting in a phagocyte dysfunction and inability to kill a spectrum of bacteria and fungi. Despite the fact that CGD patients are more susceptible to intracellular infections, visceral leishmaniasis has been reported rarely in these cases. Here, we report an uncommon case of visceral leishmaniasis in a child with CGD. An 8-yr old boy with CGD presented to the infectious disease ward, Children's Medical Center, Tehran University of Medical Sciences, Iran after the onset of 20 days fever with chronic crusted ulcer approximately 3 cm × 3cm on the left upper limb and a small ulcer measuring 0.5 cm × 0.5 cm on the right knee with moderate secretion. Bone Marrow Aspiration (BMA) and Bone Marrow Biopsies (BMB) of fragmented samples were performed and polymorphic population of hematopoietic cells, Megakaryocytes and Leishman bodies were seen. The patient was treated with meglumine antimoniate (Glucantime®) 20 mg/kg for 28 days and after partial improvement patient discharged and continue the treatment at home. Amphotericin B lipid complex (Ambisome®) (3–5 mg/kg per dose once) was administered every 3 -4 weeks for 18 months as secondary prophylaxis that was well tolerated and effective. https://ijpa.tums.ac.ir/index.php/ijpa/article/view/3226 https://ijpa.tums.ac.ir/index.php/ijpa/article/download/3226/1119