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Introduction Neuromyelitis optica spectrum disorder (NMOSD) is a rare autoimmune disease of the central nervous system which causes recurrent relapses, resulting in blindness, paralysis, and spinal cord damage. This study sought to explore the real-world burden, treatment, and cost of NMOSD in Germany using claims data. Methods Our study consisted of a retrospective analysis of two anonymized health insurance datasets covering around 9 million patients in Germany from 01/01/2013 to 31/12/2019. NMOSD patients were identified using inpatient and outpatient International Classification of Diseases, Tenth Revision (ICD-10) diagnoses of neuromyelitis optica (NMO; G36.0) and relevant symptom codes. Active periods of disease were identified based on relapse events (including hospitalizations and acute treatment); healthcare resource utilization (HCRU) and direct costs were allocated to active and inactive periods based on treatment dates. Propensity score matching was used to compare HCRU and cost outcomes among patients with and without NMOSD. Results Overall, 130 patients were identified as having NMOSD (mean age: 46.84 years; 58% female). NMOSD patients recorded 16.52 active and 348.48 inactive days per patient year (PPY). HCRU and associated costs were approximately tenfold higher during active periods than during inactive periods, with the largest share of the cost difference driven by hospitalizations (€6424.09/€259.10 per active/inactive month) and outpatient drug prescriptions (€412.83/€271.58). Direct healthcare costs incurred by patients with NMOSD (€12,913.28 PPY) were approximately threefold higher than those incurred by patients without NMOSD (€4667.66 PPY). Costs of hospitalization (€6448.32/€1937.64 PPY) and outpatient prescriptions (€3335.67/€1037.64 PPY) contributed most strongly to the difference. Conclusion Patients with NMOSD consume substantial healthcare resources and incur heavy costs during active disease phases. This study captured direct measurable healthcare costs and likely underestimates the real societal/emotional burden on patients and their families. Nevertheless, prevention of acute relapses represents one compelling strategy to minimize the economic burden of NMOSD in Germany. Supplementary Information The online version contains supplementary material available at 10.1007/s40120-021-00311-x.

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