Cost-Effectiveness Analysis of Adalimumab for the Treatment of Uveitis Associated with Juvenile Idiopathic Arthritis

Hughes, Dyfrig; Culeddu, Giovanna; Plumpton, Catrin; Wood, Eifiona; Dick, Andrew D.; Jones, Ashley P; McKay, A. J.; Williamson, Paula; Lacassagne, Sandrine; Hardwick, Ben; Hickey, Helen; Woo, Patricia; Beresford, Michael W.; Ramanan, Athimalaipet V

doi:10.1016/j.ophtha.2018.09.043

Cited by 19 publications

(41 citation statements)

References 20 publications

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“…2). The costs reported by studies from European countries [1,7,14,17,18,27] were considerably higher than those reported by other regions [4,15,20,26] (Table 1).…”

Section: Information On Jia Costsmentioning

confidence: 78%

“…1). Eleven were economic studies [1,7,[14][15][16][17][18][19][20][21][22], three retrospective observational [23][24][25] and four cohorts [4,[26][27][28].…”

Section: Selection Of Studiesmentioning

confidence: 99%

“…In 11 articles it was possible to obtain data on JIA classification (2,121 patients), being oligoarticular the most frequent (Supplementary Table 1). The follow-up was 12 months in 11 studies [1,4,7,15,17,18,20,22,[26][27][28], in the rest it was variable with a maximum of 10 years [14,16,19,21,[23][24][25] (Table 1).…”

Section: Characteristics Of Studiesmentioning

confidence: 99%

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Economic impact of Juvenile Idiopathic Arthritis: a systematic review

et al. 2021

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Background Juvenile Idiopathic Arthritis (JIA) requires complex care that generate elevated costs, which results in a high economic impact for the family. The aim of this systematic review was to collect and cluster the information currently available on healthcare costs associated with JIA after the introduction of biological therapies. Methods We comprehensively searched in MEDLINE, EMBASE, Web of Science, Scopus, and Cochrane Databases for studies from January 2000 to March 2021. Reviewers working independently and in duplicate appraised the quality and included primary studies that report total, direct and/or indirect costs related to JIA for at least one year. The costs were converted to United States dollars and an inflationary adjustment was made. Results We found 18 eligible studies including data from 6,540 patients. Total costs were reported in 10 articles, ranging from $310 USD to $44,832 USD annually. Direct costs were reported in 16 articles ($193 USD to $32,446 USD), showing a proportion of 55 to 98 % of total costs. Those costs were mostly related to medications and medical appointments. Six studies reported indirect costs ($117 USD to $12,385 USD). Four studies reported costs according to JIA category observing the highest in polyarticular JIA. Total and direct costs increased up to three times after biological therapy initiation. A high risk of reporting bias and inconsistency of the methodology used were found. Conclusion The costs of JIA are substantial, and the highest are derived from medication and medical appointments. Indirect costs of JIA are underrepresented in costs analysis.

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“…2). The costs reported by studies from European countries [1,7,14,17,18,27] were considerably higher than those reported by other regions [4,15,20,26] (Table 1).…”

Section: Information On Jia Costsmentioning

confidence: 78%

“…1). Eleven were economic studies [1,7,[14][15][16][17][18][19][20][21][22], three retrospective observational [23][24][25] and four cohorts [4,[26][27][28].…”

Section: Selection Of Studiesmentioning

confidence: 99%

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Economic impact of Juvenile Idiopathic Arthritis: a systematic review

et al. 2021

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“…Hughes et al reported that in the United Kingdom, adalimumab does not currently represent a cost-effective treatment option for the public health system. 31 Other authors, however, opposed the conclusions of the study, contesting the methodology used and emphasizing the lack of therapeutic options in cases of resistance to methotrexate, with the same clinical efficacy as adalimumab. 32 , 33 In the present review, only one study (Ramanan 2017 21 ) reported data on quality of life and the cost-effectiveness of treatment.…”

Section: Discussionmentioning

confidence: 99%

Biological Therapy in Noninfectious Pediatric Uveitis: A Systematic Review

Norcia

Kiappe

Jorge

2021

OPTH

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Purpose: Noninfectious pediatric uveitis is a potentially blinding disease often associated with systemic conditions. In cases of chronic anterior uveitis without adequate response to steroids and immunosuppressants, biological response modifiers would be viable therapeutic options. Still, evidence is lacking on the safety of the long-term use of these drugs in children. Therefore, this study aimed to evaluate the efficacy and safety of biological therapy to treat noninfectious pediatric uveitis. Methods: A systematic review was performed to identify original studies involving biological therapy for children diagnosed with noninfectious uveitis. Quality of evidence was assessed using the Grading of Recommendations, Assessment, Development, and Evaluation (GRADE) classification system. Results: Nine studies involving 526 children were eligible. Adalimumab was superior to placebo in reducing inflammatory activity (risk ratio (RR) 3.21 [95% confidence interval (CI) 1.65-6.27]; P = 0.0006; I 2 = 0%) and steroid use (RR 2.27 [95% CI 1.03-4.99]; P = 0.04; I 2 = 0%, low-certainty evidence). There was no difference between adalimumab and placebo in the occurrence of systemic adverse events (RR 2.51 [95% CI 0.74-8.54]; P = 0.14; I 2 = 48%) and local events (RR 1.15 [95% CI 0.46-2.88]; P= 0.76; I 2 = 1%). There was no difference between adalimumab and infliximab in response to treatment (RR 1.18 [95% CI 0.69-2.03]; P= 0.55; I 2 = 91%, very low-certainty evidence) and in the occurrence of adverse effects (RR 0.84 [95% CI 0.41-1.73]; P= 0.64; I 2 = 18%, low-certainty evidence). Conclusion:There is low to very-low evidence that biological therapy is effective and safe in managing noninfectious pediatric uveitis. Future large randomized trials may provide more substantial evidence to confirm these results.

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“…Re: Saeedi et al: Agreement and predictors of discordance of 6 visual field progression algorithms (Ophthalmology. 2019;126:822e828) TO THE EDITOR: We read with interest the article by Saeedi et al 1 The authors tested 6 methods to detect glaucomatous visual field (VF) progression on a large cohort of glaucomatous patients: mean deviation (MD) rate, Visual Field Index (VFI) rate, pointwise linear regression, permutation analyses of the pointwise linear regression, the Collaborative Initial Glaucoma Treatment Study scoring system, and the Advanced Glaucoma Intervention Study scoring system. They found poor to moderate intermethod agreements with only about 6% of eyes labeled as progressing by the majority of the methods.…”

mentioning

confidence: 99%

Re: Hughes et al.: Cost-effectiveness analysis of adalimumab for the treatment of uveitis associated with juvenile idiopathic arthritis (Ophthalmology. 2019;126:415-424)

et al. 2019

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TO THE EDITOR: To perform a cost-effectiveness analysis for a disease such as juvenile idiopathic arthritis (JIA)-associated uveitis is a difficult undertaking as the condition is rare and permanent visual sequelae may occur many years after the initial diagnosis. It is our belief that the methodology used by Hughes et al 1 is insufficiently robust to account for this. Their conclusions should be interpreted with a high degree of caution. To inform their cost-effectiveness analysis, Hughes et al relied heavily on data obtained from questionnaires administered to children or their proxies. The usefulness of questionnaire-based studies on patients with JIA-associated uveitis may be of limited validity. The purpose of treating these patients early and aggressively is not necessarily to "treat" impaired vision. Rather, it is to prevent (or reduce the risk of) long-term vision-threatening consequences of inflammation. These include amblyopia, macular edema, cataract, glaucoma, band keratopathy, and chronic hypotony, which can potentially cause permanent vision loss or blindness. These sequelae of inflammation may occur years later as disease activity continues and cumulative damage to the eye occurs. 2 The authors also found practical difficulties with the questionnaire-based approach with incomplete data, meaning that only 28 patients had sufficient data to calculate qualityadjusted life-years and just 3 of these patients were in the placebo arm of the trial. They commented that this required "a strong assumption of data being missing at random," an assumption that may well be flawed given the difference in the numbers between the 2 groups. The SYCAMORE trial did not provide data on the long-term outcomes of JIA-associated uveitis. 3 Hughes et al attempted to derive this information retrospectively from a cohort of patients who were under the care of a pediatric uveitis clinic, but in the description given (Table 4 in the original article), only 58% of this cohort had a diagnosis of JIA-associated uveitis. According to the published description of this cohort, 4 many of the children were already on biologic therapy, especially those with a diagnosis of JIA. Data follow-up was relatively short and only 19 patients with JIA had 10-year data. This serves to underestimate the potential effect of the disease on long-term visual outcome. A further drawback of this study, as the authors acknowledge, was that the mathematical model used was not able to consider either severe visual impairment or blindness, both of which are very real risks of undertreated JIA-associated uveitis. 2 Aside from the quality-of-life costs to the affected individual, the associated economic cost of a potentially blinding disease affecting individuals at the beginning of life is highly relevant. Health systems will look for the most suitable guidance available to inform decisions regarding the cost-effectiveness of newer treatments. For the reasons given, it is our belief that this article is unsuitable for this purpose.

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Cost-Effectiveness Analysis of Adalimumab for the Treatment of Uveitis Associated with Juvenile Idiopathic Arthritis

Cited by 19 publications

References 20 publications

Economic impact of Juvenile Idiopathic Arthritis: a systematic review

Economic impact of Juvenile Idiopathic Arthritis: a systematic review

Biological Therapy in Noninfectious Pediatric Uveitis: A Systematic Review

Re: Hughes et al.: Cost-effectiveness analysis of adalimumab for the treatment of uveitis associated with juvenile idiopathic arthritis (Ophthalmology. 2019;126:415-424)

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