Neuromyelitis Optica Spectrum Disorder: Clinical Burden and Cost of Relapses and Disease-Related Care in US Clinical Practice

Royston, Minying; Kielhorn, A; Weycker, Derek; Shaff, Melody; Houde, Linnea; Tanvir, Imran; Bhattacharyya, Shamik; Lévy, Michaël

doi:10.1007/s40120-021-00253-4

Cited by 30 publications

(31 citation statements)

References 15 publications

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“…Our study confirms findings from the USA that patients with NMOSD incur substantially higher healthcare costs than matched patients without NMOSD, and that the economic and health resource burden is the greatest during periods of active disease [ 16 , 17 ].…”

Section: Discussionsupporting

confidence: 89%

“…Our slightly more conservative detection technique used in the sensitivity analysis yielded a lower prevalence of 1.09/100,000, with higher comparability to prevalence figures found in other northern European nations, including Denmark (1.09/100,000) and Sweden (1.04/100,000) [ 21 , 22 ]. The average age of patients included in our study (base cohort/sensitivity: 46.8/46.6 years) was comparable to those observed in two US claims data studies, with respective averages of 44.9 and 46 years [ 16 , 17 ]. However, the percentage of females was marginally lower (base cohort/sensitivity: 58%/64%) than figures cited elsewhere in the literature (66–88%) [ 16 , 17 , 23 ].…”

Section: Discussionsupporting

confidence: 79%

“…In line with findings from a case control study [ 10 ] and previous claims data research [ 17 ], each 30-day period following a relapse event was defined as an active period, in order to account for the prescribed therapy course for acute attacks (normally the administration of intravenous methylprednisolone for 5 days followed by oral steroids), which warrants treatment of the patient over several days or weeks. Clusters of acute treatment codes within 30 days of one another did not result in the extension of the active period; however, all hospitalizations with a main NMO, TM, or ON code were considered active, even if the duration was longer than 30 days.…”

Section: Methodsmentioning

confidence: 99%

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Evaluating the Economic Burden of Relapses in Neuromyelitis Optica Spectrum Disorder: A Real-World Analysis Using German Claims Data

Knapp¹,

Hardtstock²,

Wilke

et al. 2021

Neurol Ther

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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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Section: Discussionsupporting

confidence: 89%

Section: Discussionsupporting

confidence: 79%

Section: Methodsmentioning

confidence: 99%

See 1 more Smart Citation

Evaluating the Economic Burden of Relapses in Neuromyelitis Optica Spectrum Disorder: A Real-World Analysis Using German Claims Data

Knapp¹,

Hardtstock²,

Wilke

et al. 2021

Neurol Ther

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“…The socioeconomic burden of relapses and long-term disease-related care is substantial. In a cohort of 1363 patients with NMOSD in the US, 47.7% experienced one or more relapses during a median follow up period of two years [52]. The average healthcare cost among patients with NMOSD was $60,599 per year compared with $8912 per year for patients without NMOSD [52].…”

Section: Treatments For Acute Attacks Of Nmosdmentioning

confidence: 99%

“…In a cohort of 1363 patients with NMOSD in the US, 47.7% experienced one or more relapses during a median follow up period of two years [52]. The average healthcare cost among patients with NMOSD was $60,599 per year compared with $8912 per year for patients without NMOSD [52]. Apart from preventive therapies to reduce relapses, prompt and efficacious treatment of acute attacks is crucial to hasten recovery, reduce neurological damage, improve functional outcomes, and lower healthcare expenditure.…”

Section: Treatments For Acute Attacks Of Nmosdmentioning

confidence: 99%

Treatment of Neuromyelitis Optica Spectrum Disorders

Chan

Lee

2021

IJMS

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Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune central nervous system (CNS) inflammatory disorder that can lead to serious disability and mortality. Females are predominantly affected, including those within the reproductive age. Most patients develop relapsing attacks of optic neuritis; longitudinally extensive transverse myelitis; and encephalitis, especially brainstem encephalitis. The majority of NMOSD patients are seropositive for IgG autoantibodies against the water channel protein aquaporin-4 (AQP4-IgG), reflecting underlying aquaporin-4 autoimmunity. Histological findings of the affected CNS tissues of patients from in-vitro and in-vivo studies support that AQP4-IgG is directly pathogenic in NMOSD. It is believed that the binding of AQP4-IgG to CNS aquaporin-4 (abundantly expressed at the endfoot processes of astrocytes) triggers astrocytopathy and neuroinflammation, resulting in acute attacks. These attacks of neuroinflammation can lead to pathologies, including aquaporin-4 loss, astrocytic activation, injury and loss, glutamate excitotoxicity, microglial activation, neuroinflammation, demyelination, and neuronal injury, via both complement-dependent and complement-independent pathophysiological mechanisms. With the increased understanding of these mechanisms underlying this serious autoimmune astrocytopathy, effective treatments for both active attacks and long-term immunosuppression to prevent relapses in NMOSD are increasingly available based on the evidence from retrospective observational data and prospective clinical trials. Knowledge on the indications and potential side effects of these medications are essential for a clear evaluation of the potential benefits and risks to NMOSD patients in a personalized manner. Special issues such as pregnancy and the coexistence of other autoimmune diseases require additional concern and meticulous care. Future directions include the identification of clinically useful biomarkers for the prediction of relapse and monitoring of the therapeutic response, as well as the development of effective medications with minimal side effects, especially opportunistic infections complicated by long-term immunosuppression.

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Characterizing mortality in patients with AQP4‐Ab+ neuromyelitis optica spectrum disorder

Francis,

Gibbons,

et al. 2024

Ann Clin Transl Neurol

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Neuromyelitis optica spectrum disorder is an autoimmune disease, causing severe disability due to relapses, but recent mortality data are limited. Among 396 patients seropositive for anti‐aquaporin‐4 antibody from 2014 to 2020 in the United Kingdom, 39 deaths occurred: 19 (48.7%) were unrelated to disease; 14 (35.9%) were severe disability‐ or relapse‐related; and 4 (10.3%) were attributed to malignancy/infection. Mean annual mortality was 1.92% versus 0.63% in the matched population. The standardized mortality ratio was 3.04 (95% confidence interval 1.67–5.30) with 1.29% excess mortality per year in patients. Median Expanded Disability Status Scale before death was 7.0. Results highlight the importance of preventing relapses that drive disability.

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Neuromyelitis Optica Spectrum Disorder: Clinical Burden and Cost of Relapses and Disease-Related Care in US Clinical Practice

Cited by 30 publications

References 15 publications

Evaluating the Economic Burden of Relapses in Neuromyelitis Optica Spectrum Disorder: A Real-World Analysis Using German Claims Data

Evaluating the Economic Burden of Relapses in Neuromyelitis Optica Spectrum Disorder: A Real-World Analysis Using German Claims Data

Treatment of Neuromyelitis Optica Spectrum Disorders

Characterizing mortality in patients with AQP4‐Ab+ neuromyelitis optica spectrum disorder

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