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01.12.2014 | Research | Ausgabe 1/2014 Open Access

Orphanet Journal of Rare Diseases 1/2014

The economic impact of Marfan syndrome: a non-experimental, retrospective, population-based matched cohort study

Zeitschrift:
Orphanet Journal of Rare Diseases > Ausgabe 1/2014
Autoren:
Dmitrij Achelrod, Carl Rudolf Blankart, Roland Linder, Yskert von Kodolitsch, Tom Stargardt
Wichtige Hinweise

Electronic supplementary material

The online version of this article (doi:10.​1186/​1750-1172-9-90) contains supplementary material, which is available to authorized users.

Competing interests

RL is employed by Techniker Krankenkasse, a large German sickness fund, and may have potential competing interests according to the guidelines of the International Committee of Medical Journal Editors. YK is the chairman of the non-profit patient advocacy organisation ‘Marfan Hilfe e.V.’. DA, CRB, YK and TS declare that they have no competing interests.

Authors’ contributions

CRB and TS collected the data. RL participated in the coordination of the data collection. DA, CRB, and TS developed the design of the study and performed the statistical analysis. DA, CRB, RL, YK and TS drafted the manuscript. All authors read and approved the final manuscript.

Abstract

Background

Marfan syndrome is a rare disease of the connective tissues, affecting multiple organ systems. Elevated morbidity and mortality in these patients raises the issue of costs for sickness funds and society. To date, there has been no study analysing the costs of Marfan syndrome from a sickness fund and societal perspective.

Objective

To estimate excess health resource utilisation, direct (non-)medical and indirect costs attributable to Marfan syndrome from a healthcare payer and a societal perspective in Germany in 2008.

Methods

A retrospective matched cohort study design is applied, using claims data. For isolating the causal effect of Marfan syndrome on excess costs, a genetic matching algorithm was used to reduce differences in observable characteristics between Marfan syndrome patients and the control group. 892 patients diagnosed with Marfan syndrome (ICD-10 Q87.4) were matched from a pool of 26,645 control individuals. After matching, we compared health resource utilisation and costs.

Results

From the sickness fund perspective, an average Marfan syndrome patient generates excess annual costs of €2496 compared with a control individual. From the societal perspective, excess annual costs amount to €15,728. For the sickness fund, the strongest cost drivers are inpatient treatment and care by non-physicians. From the sickness fund perspective, the third (25–41 years) and first (0–16 years) age quartiles reveal the greatest surplus in total costs. Marfan syndrome patients have 39% more physician contacts, a 153% longer average length of hospital stay, 119% more inpatient stays, 33% more prescriptions, 236% more medical imaging and 20% higher average prescription costs than control individuals. Depending on the prevalence, the economic impact from the sickness fund perspective ranges between €24.0 million and €61.4 million, whereas the societal economic impact extends from €151.3 million to €386.9 million.

Conclusions

Relative to its low frequency, Marfan syndrome requires high healthcare expenditure. Not only the high costs of Marfan syndrome but also its burden on patients’ lives call for more awareness from policy-makers, physicians and clinical researchers. Consequently, the diagnosis and treatment of Marfan syndrome should begin as soon as possible in order to prevent disease complications, early mortality and substantial healthcare expenditure.
Zusatzmaterial
Additional file 1: Table A1: Elixhauser comorbidity groups and pharmacy-based metrics before and after matching with balance statistics. Table A2. Cost estimates in € for Marfan and controls, by age quartiles and analysis perspective (per capita in 2008). Table A3. Sensitivity analysis of estimated outcomes with Rosenbaum bounds at different magnitudes of hidden bias (1.0≤Γ≤3.0). (XLSX 26 KB)
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Authors’ original file for figure 1
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Authors’ original file for figure 2
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Literatur
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