Tehran University of Medical Sciences Iranian Journal of Parasitology 1735-7020 15 4 2020 12 07 608 614 Darya POKUTNAYA Department of Epidemiology, School of Public Health, University of Pittsburgh, Pittsburgh, PA, USA Mohammad Reza SHIRZADI Center for Research of Endemic Parasites of Iran, Tehran University of Medical Sciences, Tehran, Iran AND Center for Communicable Diseases Control, Ministry of Health and Medical Education, Tehran, Iran Elham SALARI Biocontrol and Insect Pathology Laboratory, University of Applied Science and Technology, Kerman, Iran Goudarz MOLAEI Department of Environmental Sciences, Center for Vector Biology & Zoonotic Diseases, The Connecticut Agricultural Experiment Station, New Haven, Connecticut, USA AND The Northeast Center for Excellence in Vector-Borne Diseases, The Connecticut Agricultural Experiment Station, New Haven, Connecticut, USA AND Department of Epidemiology of Microbial Diseases, Yale School of Public Health, New Haven, Connecticut, USA 2020 12 07 Cutaneous leishmaniasis (CL) is an emergent public health concern, particularly in tropical and subtropical regions. Reports of pregnancy complications are scarce; however, as the endemic range of CL expands in Iran, there is concern of possible detrimental effects on fetal development amongst infected mothers through placental transmission of the parasite or enhanced maternal immune responses. We herein describe the first known case of persistent anthroponotic CL, plausibly responsible for pregnancy complications, preterm birth, and neonatal death in a healthy Iranian primigravida woman. Diagnosis was based on physical examinations of the lesions on the back of both calves of the patient and laboratory analyses including direct smear, culture, and PCR. During active CL infection, the patient gave birth to a premature female neonate who passed three days post-delivery due to immature lung development and subsequent respiratory distress syndrome. This report highlights the challenges associated with CL infection during pregnancy, exacerbation of lesions, and subsequent complications. https://ijpa.tums.ac.ir/index.php/ijpa/article/view/3190 https://ijpa.tums.ac.ir/index.php/ijpa/article/download/3190/1099