Tehran University of Medical Sciences Iranian Journal of Parasitology 1735-7020 14 3 2019 09 22 472 476 Mahdi MOSAYEBI Department of Medical Parasitology and Mycology, School of Medicine, Arak University of Medical Sciences, Arak, Iran AND Health Center Laboratory, University of Medical Sciences, Arak, Iran Mehdi MOHEBALI Department of Medical Parasitology and Mycology, School of Public Health, Tehran University of Medical Sciences, Tehran, Iran AND Research Center for Endemic Parasites of Iran, Tehran University of Medical Sciences, Tehran, Iran Aliasghar FARAZI Department of Infectious Diseases, School of Medicine, Arak University of Medical Sciences, Arak, Iran Mohammad Reza SHIRZADI Communicable Diseases Management Center, Ministry of Health and Medical Education, Tehran, Iran Davood AKHLAGHI Health Center Laboratory, University of Medical Sciences, Arak, Iran Reza HAJHOSSEIN Department of Medical Parasitology and Mycology, School of Medicine, Arak University of Medical Sciences, Arak, Iran Samira ELIKAEE Department of Medical Parasitology and Mycology, School of Public Health, Tehran University of Medical Sciences, Tehran, Iran 2018 10 06 2019 01 14 Cutaneous leishmaniasis (CL) is one of the prevalent parasitic diseases in Iran principally caused by two species, Leishmania major and L. tropica. Here, we present a rare case of a congenital form of CL around the glans penis from the central part of Iran in 2017. A 24-yr-old male patient from the central part of Iran presented with biennial ulceration of the glans penis. Diagnostic methods included physical and preclinical examination, microscopic observation, leishmanin skin test (LST), and serological tests including direct agglutination test (DAT). Nested PCR and sequencing analysis were used on the positive smears for confirmation of CL and Leishmania species identification. The preclinical results were normal, and no anti-Leishmania antibodies were detected in the peripheral blood of the patient using DAT. In abdominal ultrasonography, the spleen and liver size were normal. LST was positive (≥5 mm) after 72 h, and a few amastigote forms of Leishmania sp. were demonstrated under light microscopy. L. major was confirmed using nested PCR and sequencing analysis. The patient responded to oral administration of miltefosine (2.5 mg/kg/d) for 28 days. To the best of our knowledge, genital CL due to L. major has not been previously reported from Iran. https://ijpa.tums.ac.ir/index.php/ijpa/article/view/2328