OBJECTIVES: To estimate the annual per patient cost of Amyotrophic Lateral Sclerosis (ALS) in Greece from a societal point of view. METHODS: Data on direct costs (medications, laboratory/imaging tests, consultations, hospitalizations) were obtained through retrospective chart review of a sample of patients followed-up in Aeginition University Hospital, a reference centre for ALS in Greece. Eligible patients were those that visited the ALS clinic in the previous 6 months and had fully recorded data for the previous year. Patients were also personally interviewed, following consent, based on a strictly-structured questionnaire, with an aim to record indirect costs incurred in the previous year (work absenteeism, professional home help, walking aids). Unit prices for health-resource use were the official NHS prices. Work loss and home help were costed with the hourly rate of the basic salary, in order to obtain a conservative approach. Costs are reported in year 2011 Euros RESULTS: The sample (N=34) was 53% female with an average age of 61.6 years. Total average annual per patient cost was 7450.6€ (standard deviation: 6423€), out of which 4136.3€ (s.d. 1,350€) were direct and 3314.2€ (s.d. 6190€) were indirect expenditure. Medications accounted for 32.7% of the total cost, followed by professional home help (24.4%), work absenteeism (17.7%) and hospitalizations (9.2%). Women had a significantly higher average cost than men (10,004€ vs. 4,347€, p<0.05), mostly as a result of indirect expenditures from productivity loss. Age >65 did not have a significant impact on outcomes due to the substitution of productivity losses (<65) by home help, for patients >65. CONCLUSIONS: ALS entails a significant per patient economic burden in societal terms. Cost-of-illness data, even for rare diseases, provide important inputs for the decision-making process in health as well as for awareness purposes.