AbstractImportanceStargardt disease (SD) is the most common juvenile macular degeneration and a leading cause of uncorrectable childhood blindness. The progressive and incurable nature of this chronic condition entails a long-term financial burden on affected individuals. The economic costs of SD have not been characterized in detail.ObjectiveTo estimate the direct healthcare cost of SD.DesignCross-sectional analysis of healthcare claims.ParticipantsPatients with an ICD-9 diagnosis code of SD, non-exudative age-related macular degeneration (AMD), or bilateral sensorineural hearing loss (SHL).MethodsOutpatient administrative claims data from the IBM® MarketScan® Commercial Claims and Encounters Database from 2010 to 2014 were analyzed.Main Outcome MeasureAnnual per-patient direct healthcare cost.ResultsA total of 472,428 patients were analyzed (5,015 SD, 369,750 SHL and 97,663 AMD patients respectively). The payment per year of insurance coverage for SD (median: $105.58, IQR: $50.53-$218.71) was higher than that of SHL (median: $51.01, IQR: $25.66-$121.66, p <0.001) and AMD (median: $76.20, IQR: $38.00-$164.86, p <0.001). When adjusted for covariates, the annual payment for SD was $47.83 higher than SHL (p<0.001) and $17.34 higher than AMD (p<0.001).Conclusions and RelevanceThere is a significant direct healthcare cost associated with SD. The annual per-patient cost of SD was higher than SHL, another condition that causes sensory impairment in people of all ages, and nonexudative AMD which causes a similar pattern of visual loss that typically begins later in life. The total lifetime per-patient cost of SD may exceed that of nonexudative AMD.