BACKGROUND: Visceral leishmaniasis (VL) is an important public health problem, which mainly affects the poor rural dwelling communities in Low- and Middle-Income Countries. However, little is known about the health and economic burdens of this disease in East Africa, including Ethiopia. The aim of this study was to assess the household level economic burden of VL among affected communities in Tigray, Northern Ethiopia. METHODS: Between April and August 2020, a cross-sectional household survey was conducted on 96 patients who had been treated for VL within 12 months prior to the survey, in six districts of Tigray. Data on households' health seeking behavior, direct and indirect costs and coping strategies were collected using a structured questionnaire and the responses were analyzed using SPSS software. RESULTS: Most (82%) of the patients surveyed were males and the majority (74%) of them were between 16 and 30 years of age. The education level of participants was very low: over 33% had not received any form of education; 48% of patients were farmers dependent on subsistence agriculture and about 32% were daily laborers. Just under half of household families (46%) resided in "poor houses" with structures made from entirely local materials. Forty-one percent of patients from the surveyed households had traveled 48 to 72 kilometers to reach VL treatment hospitals. The median total household cost for one VL episode was estimated to be US$ 214. This is equated to 18% of the mean total annual household income or 72.5% of annual per capita income of the study population. More than 80% of the households surveyed incurred catastrophic costs of VL, where this is defined as exceeding 10% of annual household income. The median delay between the onset of symptoms and arrival at a care provider hospital was 37 days; once the patient arrived at hospital, the median delay during diagnosis was 3 days. Direct and indirect costs represented 44% and 56% of the total costs incurred, respectively. To cope with VL treatment costs, 43% of the households used more than one coping strategy: 48% took out loans, 43% sold livestock and 31% of households mobilized cash savings. CONCLUSIONS: VL in Tigray is concentrated among young males with low educational background and mostly engaged in subsistence economic activities. Despite the free diagnostic and treatment provisions that were available at public hospitals at the time of the study, our work shows that the household economic burden of the disease had significant impact among VL-affected communities in Tigray. Initiating community awareness towards prevention, early treatment seeking and decentralization of VL treatment centers are strongly recommended. In addition, we recommend efforts to reduce household treatment costs through transport and food provisions for patients (and their accompanying carers where possible) or through cash reimbursement for patients who complete treatment at public hospitals, in order to reduce the barriers to seeking treatment for this life-threatening disease.