C. Baldini

G. Turchetti

I. Palla

L. Trieste

L.S. D'Angiolella

M. Mosca

R. Talarico

TRIESTE, LEOPOLDO

PALLA, Ilaria

TURCHETTI, Giuseppe

L. S. D'Angiolella

Archivio della ricerca della Scuola Superiore Sant'Anna

S-1541Istituto di Management, Scuola Superiore Sant’Anna, Pisa, Italy; 2Rheumatology Unit, Department of  Internal Medicine, University of Pisa, Italy;3Fondazione Charta, Milan, Italy. Leopoldo Trieste, PhDIlaria PallaChiara Baldini, MD, PhDRosaria Talarico, MD, PhDLucia D’AngiolellaMarta Mosca, MD, PhDGiuseppe Turchetti, PhD, Fulbright ScholarPlease address correspondence to: Prof. Giuseppe Turchetti, Istituto di Management, Scuola Superiore Sant’Anna, Piazza Martiri della Libertà 33, 56127 Pisa, Italy.E-mail: g.turchetti@sssup.itReceived and accepted on October 1, 2012.Clin Exp Rheumatol 2012; 30 (Suppl. 73): S154-S156.© Copyright CLINICAL AND EXPERIMENTAL RHEUMATOLOGY 2012.Key words: systemic vasculitis, direct costs, indirect costs. Competing interests: none declared.ABSTRACT This article attempts to perform an evaluation of the state of the art of the economic and societal burden of sys-temic vasculitis (VAs). Due to the rar-ity of these diseases and their variable clinical picture, few data are available in the literature on their health eco-nomic issues, and only some papers have been published that marginally examine the problem. Since VAs are severe conditions with a high medical and societal impact and determine high healthcare resource con-sumption, studies able to deﬁne societal, quality of life and economic burden of these pathologies are needed. Policy makers, private and public organisa-tions involved in the care of VAs need data to programme future investment or make cost-effectiveness analysis for in-troducing new drugs or protocols. IntroductionSystemic vasculitis (VAs) are an het-erogeneous group of diseases, often characterised by a severe course. It may therefore be argued that, in addition to the impact on patients outcome and prognosis, these conditions, as many other rheumatic diseases, may have a high impact on the healthcare systems, the health care costs, and the society (1-3).Aim of the present review is to exam-ine the last decade literature studies on the issue of healthcare costs in VAs.The review agrees the recommenda-tions of the Centre for Reviews and Dissemination (4) and of the Cochrane Collaboration (5), thereby using an established rigorous and reproducible methodology. A protocol was devel-oped to deﬁne review questions.MethodsPublished studies in English were searched using the main electronic database, PubMed MEDLINE. The search was performed for the period January 2002–September 19, 2012. The search strategy is as follows: ((“economics”[Subheading] OR “economics”[All Fields] OR “cost”[All Fields] OR “costs and cost analysis”[MeSH Terms] OR (“costs”[All Fields] AND “cost”[All Fields] AND “analysis”[All Fields]) OR “costs and cost analysis”[All Fields]) AND (“vasculitis”[MeSH Terms] OR “vasculitis”[All Fields])) AND ((“2002/01/01”[PDAT]: “2012/09/19” [PDAT]) AND “humans” [MeSH Terms] AND English[lang] AND “adult”[MeSH Terms]).The publications were assessed for in-clusion by a 3-step process: i. titles and abstracts of all identiﬁed studies were assessed by one reviewer and checked by a second reviewer; ii. full texts of relevant articles were then obtained and inclusion criteria applied independently by two reviewers. Possible discords be-tween reviewers were resolved by con-sensus; iii. data were extracted by one reviewer and then checked by a second reviewer.Inclusion criteriaIn the study protocol the reviewers se-lected publications from the mentioned database as follows:Period: Jan. 2002–Sep. 19, 2012Language: EnglishStudies: all articles related to economic analysisPatients: adult ≥18 Outcomes: direct costs, indirect costsExclusion criteriaNot published in English and all pub-lications before 2002, conferences proceedings, case reports, reviews, systematic reviews, letters and com-mentaries were excluded. ResultsUp to September 19 2012, 35 articles were extracted, but only 3 papers were considered relevant to this review (Fig. 1) (6-8). Systemic vasculitis: how little we know about their societal and economic burdenL. Trieste1, I. Palla1, C. Baldini2, R. Talarico2, L. D’Angiolella3, M. Mosca2, G. Turchetti1 S-155The economic burden of systemic vasculitis / L. Trieste et al.Excluded articles had no indications of societal and/or economic impacts or considered only the impact of some therapies. Based on these results, a sys-tematic literature review was not possi-ble and therefore data are summarised below as a descriptive review.The societal and economic impact of VAs  Single studies have examined the soci-etal impact of VAs. Reinhold-Keller et al. (8) registered a very high frequency of hospitalisation and visits for Wege-ner’s granulomatosis (WG) patients. More than the half of WG patients had been hospitalised in the year before the study. In extreme cases these authors registered one visit a week. As far as the employment status was concerned, 27% of patients were un-employed because of the disease, while employed patients reported a mean of 14 workdays lost per year because of WG. In this study, having WG diagnosis and being unemployed was associated with reduction of social and physical func-tioning.Few studies speciﬁcally addressing costs in VAs are available. Krulichova et al. (6) estimate the amount of re-sources used and direct medical costs associated to active versus inactive Takayasu’s arteritis (TA) in Italy, in a multicentre and prospective study. The study reports €5054.3 vs. €1328.4 of mean annual costs per TA active and TA inactive patients, respectively. These costs are mainly generated by drug therapy (22.0% of the total costs) and hospitalisation (44.8% of total costs), measured using Diagnosis Re-lated Groups (DRGs). The direct and indirect costs associated to Behçet’s syndrome (BS) are  report-ed by Sut et al. (7). The study includes 119 Turkish BS patients  divided into four groups based on the presence of ocular, vascular, neurological and mu-cocutaneous-joint involvement. Direct annual per patient costs ($2203 on average) fall in a interval with a left-bound of $973 for mucocutaneous-joint patients’ subgroup and a right-bound of $2727 for the ocular patients’ sub-group. Indirect annual per patient costs are $1023, on average. Highest costs have been registered within the neurological subgroup ($2190) and the lowest cost within the mucocutaneous-joint pa-tients’ subgroup. As a result, the study reports $3226 of total annual per patient BS related costs with $5005 for the neurological sub-group, $1280, $679 and $207 within the ocular, vascular and mucocutaneous-joint subgroups, respectively.  These results should be treated with caution because of the very high standard de-viations reported. Discussion and conclusions The paucity of studies on direct and in-direct costs of VAs made a systematic review not possible.The few studies assessing direct costs suggest that VAs determine high costs related with their severity, the need for frequent hospitalisation and costly pro-cedures. Contrarily to other rheumatic diseases as far as indirect costs are concerned, although many data are available in the literature on outcomes and quality of life in these conditions (8-16), no de-tailed analysis are available on the con-sequences of a poor quality of life on employment, salaries and productivity.The lack of a consolidated literature on this theme is a good opportunity to de-sign and conduct ad hoc studies – also deﬁning the criteria and the issues that must be included in the analysis – in order to obtain statistically signiﬁcant and cross countries comparable results useful also for deriving health policy implications (17, 18). References   1. CARPENTER DM, MEADOR AE, ESTAD EA, HOGAN SL, DE VELLIS RF: The impact of vasculitis on patients’ social participation and friendships. Clin Exp Rheumatol 2012; 30: S15-S21.  2. TALARICO R, BALDINI C, DELLA ROSSA A et al.: Large- and small-vessel vasculitis: a criti-cal digest of the 2010-2011 literature. Clin Exp Rheumatol 2012; 30 (Suppl 70): S130-8. 3. MOSCA M, BOUMPAS D, BRUCE IN et al.: Treat-to-target in systemic lupus erythemato-sus : where are we today? Clin Exp Rheuma-tol 2012; 30 (Suppl. 73): S112-S115.  4. CENTRE FOR REVIEWS AND DISSEMINATION: CRD’s Guidance for Undertaking Reviews in Health Care, 2008, University of York.  5. Cochrane Handbook for Systematic Reviews of interventions, Edited by HIGGINS JPT and GREEN S, 2008, Wiley-Blackwell.  6. KRULICHOVA I, GAMBA S, RICCI E, GARAT-TINI L: Direct medical costs of monitoring and treating patients with Takayasu arteritis in Italy. Eu J Health Econ 2004; 49: 330-4.   7. SUT N, SEYAHI E, YURDAKUL S, SENOCAK M, YAZICI H: A cost analysis of Behçet’s syn-drome in Turkey. Rheumatology 2007; 46: 678-82.Fig. 1. PRISMA ﬂow diagram for cost studies in vasculitisS-156The economic burden of systemic vasculitis / L. Trieste et al.  8. REINHOLD-KELLER E, HERLYN K, WAGNER-BASTMEYER R et al.: Effect of Wegener’s Granulomatosis on work disability, need for medical care, and quality of life in patients younger than 40 years at diagnosis. Arthritis Rheum (Arthritis Care Res) 2002; 47: 320-5.   9. ABULARRAGE CJ, SLIDELL MB, SIDAWY AN, KREISHMAN P, AMDUR RL, ARORA S:  Qual-ity of life of patients with Takayasu’s arteri-tis. J Vasc Surg 2008; 131-7.  10. ERTAM I, KITAPCIOGLU G, AKSU K et al.: Quality of life and its relation with disease severity in Behçet’s disease. Clin Exp Rheu-matol 2009; 27: S18-S22.  11. TALARICO R, D’ASCANIO A, FIGUS M et al.: Behçet’s disease: features of neurological in-volvement in a dedicated centre in Italy. Clin Exp Rheumatol 2012 Sep 25 [Epub].12. FURNERI G, MANTOVANI LG, BELISARI A et al.: Systematic literature review on economic implications and pharmacoeconomic issues of rheumatoid arthritis. Clin Exp Rheumatol 2012; 30 (Suppl. 73): S72-S84.13. TURCHETTI G, YAZDANY J, PALLA I, YELIN E, MOSCA M: Systemic lupus erythematosus and the economic perspective: a systematic litera-ture review and points to consider. Clin Exp Rheumatol 2012; 30 (Suppl. 73): S117-S123.14. PALLA I, TRIESTE L, TANI C et al.: A system-atic literature review of the economic impact of ankylosing spondylitis. Clin Exp Rheuma-tol 2012; 30 (Suppl. 73): S136-S141.15. TRIESTE L, PALLA I, FUSCO F et al.: The eco-nomic impact of gout: systematic literature review. Clin Exp Rheumatol 2012; 30 (Suppl. 73): S145-S148.16. CORTESI PA, SCALONE L, D’ANGIOLELLA L et al.: Systematic literature review on eco-nomic implications and pharmacoeconomic issues of psoriatic arthritis. Clin Exp Rheu-matol 2012; 30 (Suppl. 73): S126-S131.17. TURCHETTI, G, SPADONI E, GEISLER E: Health technology assessment. Evaluation of biomedical innovative technologies. IEEE Engineering in Medicine and Biology Maga-zine 2010; 29: 70-6.18. TURCHETTI G, SCALONE L, DELLA CASA AL-BERIGHI O et al.: The rationale of pharmac-oeconomic analysis in rheumatologic indica-tions. Clin Exp Rheumatol 2012; 30 (Suppl. 73): S64-S71.

Systemic vasculitis: how little we know about their societal and economic burden

Systemic vasculitis: how little we know about their societal and economic burden

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