Cutaneous leishmaniasis (CL), a neglected tropical disease (NTD), is a major public health problem in Yemen with widespread distribution in rural areas. Evaluating the knowledge and understanding of people’s beliefs towards the disease is essential to the implementation of effective control measures. This study aims to assess the knowledge, attitudes, practices (KAP) and treatment-seeking behaviour concerning CL among rural populations in the western highlands of Yemen. A community-based cross-sectional study was conducted among 289 household heads in four rural areas of the Utmah District. Data were collected using a pre-tested questionnaire. All the participants had heard about the disease; however, only 9.3% attributed it to sandflies. Nearly half (48.1%) of the participants could not mention any preventive measures for CL, and nearly two-thirds (65.4%) could not do so for sandflies. The overall ‘good’ knowledge about CL was found to be 51.2%, and it was 33.9% for sandflies. The participants’ attitude and prevention practices towards CL were not satisfactory, as only 38.1% and 16.3% had a positive attitude and good CL-related prevention practices, respectively. Moreover, 45.7% believed CL to be a stigmatising disease, and 50% had used traditional remedies to treat suspected CL lesions. Multivariate analyses showed that age, sex, presence of CL-confirmed cases in the same household, residency, occupation and monthly household income were the significant predictors associated with KAP concerning CL among the participants. The findings support an urgent need for integrated health education and community mobilisation interventions to improve awareness of these vulnerable populations about this devastating disease.