Background Cutaneous leishmaniasis (CL) is one of the health problems of many tropical and subtropical regions and is endemic in many parts of Iran especially. Cutaneous leishmaniasis not only is a health problem but also is a social and cultural problem in Iran. Education about health for high risk population seems to have a critical role in prevention of leishmaniasis and therefore evaluation of knowledge, attitude and performance are of importance. For the success of prevention and control programs of any disease, the most important prerequisite is community participation. Program implementers need to understand the disease-related knowledge, attitude, and practices (KAP) of the community, because these are the important determinants of community participation. There are no data from Iran focusing on these aspects, and thus this study presents the information on KAP related to cutaneous leishmaniasis (C.L) in Iran. Methods In this study 166 mothers that had at least one child with CL were studied. Data collection was performed using questionnaires that were designed by epidemiologist and were analyzed using SPSS software. Results The mean of KAP score was 15.7 ± 1.6 (range of 11.5–19). 48 mothers (28.9%) had weak KAP, 79 mothers (47.6%) had average KAP and 39 mothers (23.5%) had excellent KAP. Conclusion According to our results, about 28.9% the mothers had inappropriate KAP score about leishmaniasis highlighting the fact that at least one third of the Isfahan population needs practical education about combating with leishamaniasis. To close the gap between the knowledge and practice of the mothers, face to face education and use of instructional aides are recommended.