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Published in: Journal of Neurology 10/2011

01-10-2011 | Original Communication

Cost-of-illness in multiple system atrophy and progressive supranuclear palsy

Authors: Yaroslav Winter, Maria Stamelou, Nicole Cabanel, Friedericke Sixel-Döring, Karla Eggert, Günter U. Höglinger, Birgit Herting, Thomas Klockgether, Heinz Reichmann, Wolfgang H. Oertel, Richard Dodel, Annika E. Spottke

Published in: Journal of Neurology | Issue 10/2011

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Abstract

Multiple system atrophy and progressive supranuclear palsy are disabling neurodegenerative disorders, also known as atypical parkinsonian syndromes. Currently, no health economic evaluations of these diseases are available. The objective of this study was to evaluate disease-related costs in German patients with multiple system atrophy and progressive supranuclear palsy and to identify cost-driving factors. We recruited 101 consecutive patients with multiple system atrophy (n = 54) and progressive supranuclear palsy (n = 47) in four German specialised movement disorder clinics. The health economic data were collected using comprehensive health economic questionnaires (“bottom-up” approach). Costs were calculated from the societal perspective in 2010 Euros. Independent cost-driving factors were identified in multiple regression analysis. The total semi-annual costs of atypical parkinsonian syndromes were EUR 16,670 (95% CI: 13,470–21,850). Direct costs accounted for 73% (inpatient care 31%, special equipment 24%, copayments of patients 21%, others 24%) and indirect costs for 27% of total costs. The economic burden imposed on patients by atypical parkinsonian syndromes accounted for 36% of their income. Independent cost-driving factors were younger age, disease severity, living without a partner and depression. The disease-related costs of atypical parkinsonian syndromes in Germany are high and above the costs reported for idiopathic Parkinson’s disease. Disease-specific patterns of cost distributions in atypical parkinsonian syndromes and independent cost-drivers should be considered in future health economic evaluations and healthcare programs. The early diagnosis and treatment of depression in patients with atypical parkinsonian syndromes as well as programs aimed to improve social support will reduce disease-related costs.
Literature
2.
go back to reference Barber JA, Thompson SG (2000) Analysis of cost data in randomized trials: an application of the non-parametric bootstrap. Stat Med 19:3219–3236PubMedCrossRef Barber JA, Thompson SG (2000) Analysis of cost data in randomized trials: an application of the non-parametric bootstrap. Stat Med 19:3219–3236PubMedCrossRef
3.
go back to reference Beck A, Ward CH, Mendelson M, Mock J, Erbaugh J (1961) An inventory for measuring depression. Arch Ger Psychiatry 4:561–567 Beck A, Ward CH, Mendelson M, Mock J, Erbaugh J (1961) An inventory for measuring depression. Arch Ger Psychiatry 4:561–567
4.
go back to reference Brazier J, Jones N, Kind P (1993) Testing the validity of the EuroQoL and comparing it with the SF-36 health survey questionnaire. Qual Life Res 2:169–180PubMedCrossRef Brazier J, Jones N, Kind P (1993) Testing the validity of the EuroQoL and comparing it with the SF-36 health survey questionnaire. Qual Life Res 2:169–180PubMedCrossRef
5.
go back to reference Coelho M, Marti MJ, Tolosa E, Ferreira JJ, Valldeoriola F, Rosa M, Sampaio C (2010) Late-stage Parkinson’s disease: the Barcelona and Lisbon cohort. J Neurol 257:1524–1532PubMedCrossRef Coelho M, Marti MJ, Tolosa E, Ferreira JJ, Valldeoriola F, Rosa M, Sampaio C (2010) Late-stage Parkinson’s disease: the Barcelona and Lisbon cohort. J Neurol 257:1524–1532PubMedCrossRef
6.
go back to reference Colosimo C, Tiple D, Wenning GK (2005) Management of multiple system atrophy: state of the art. J Neural Transm 112:1695–1704PubMedCrossRef Colosimo C, Tiple D, Wenning GK (2005) Management of multiple system atrophy: state of the art. J Neural Transm 112:1695–1704PubMedCrossRef
7.
go back to reference Deutscher Ärzteverlag (2006) Einheitlicher Bewertungsmasstab. Deutscher Arzteverlag, Köln Deutscher Ärzteverlag (2006) Einheitlicher Bewertungsmasstab. Deutscher Arzteverlag, Köln
8.
go back to reference Dorsey ER, Constantinescu R, Thompson JP, Biglan KM, Holloway RG, Kieburtz K, Marshall FJ, Ravina BM, Schifitto G, Siderowf A, Tanner CM (2007) Projected number of people with Parkinson disease in the most populous nations, 2005 through 2030. Neurology 68(5):384–386PubMedCrossRef Dorsey ER, Constantinescu R, Thompson JP, Biglan KM, Holloway RG, Kieburtz K, Marshall FJ, Ravina BM, Schifitto G, Siderowf A, Tanner CM (2007) Projected number of people with Parkinson disease in the most populous nations, 2005 through 2030. Neurology 68(5):384–386PubMedCrossRef
9.
go back to reference Efron B, Tibshirani RJ (1993) An introduction to the bootstrap. Chapman and Hall, London Efron B, Tibshirani RJ (1993) An introduction to the bootstrap. Chapman and Hall, London
10.
go back to reference Findley L, Aujla M, Bain PG, Baker M, Beech C, Bowman C, Holmes J, Kingdom WK, MacMahon DG, Peto V, Playfer JR (2003) Direct economic impact of Parkinson’s disease: a research survey in the United Kingdom. Mov Disord 18:1139–1145PubMedCrossRef Findley L, Aujla M, Bain PG, Baker M, Beech C, Bowman C, Holmes J, Kingdom WK, MacMahon DG, Peto V, Playfer JR (2003) Direct economic impact of Parkinson’s disease: a research survey in the United Kingdom. Mov Disord 18:1139–1145PubMedCrossRef
11.
go back to reference Folstein MF, Folstein SE, McHugh PR (1975) “Mini-mental state”: a practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res 12(3):189–198PubMedCrossRef Folstein MF, Folstein SE, McHugh PR (1975) “Mini-mental state”: a practical method for grading the cognitive state of patients for the clinician. J Psychiatr Res 12(3):189–198PubMedCrossRef
12.
go back to reference Gilman S, Low PA, Quinn N, Albanese A, Ben-Shlomo Y, Fowler CJ, Kaufmann H, Klockgether T, Lang AE, Lantos PL, Litvan I, Mathias CJ, Oliver E, Robertson D, Schatz I, Wenning GK (1999) Consensus statement on the diagnosis of multiple system atrophy. J Neurol Sci 163:94–98PubMedCrossRef Gilman S, Low PA, Quinn N, Albanese A, Ben-Shlomo Y, Fowler CJ, Kaufmann H, Klockgether T, Lang AE, Lantos PL, Litvan I, Mathias CJ, Oliver E, Robertson D, Schatz I, Wenning GK (1999) Consensus statement on the diagnosis of multiple system atrophy. J Neurol Sci 163:94–98PubMedCrossRef
13.
go back to reference Hagell P, Nordling S, Reimer J, Grabowski M, Persson U (2002) Resource use and costs in a Swedish cohort of patients with Parkinson’s disease. Mov Disord 17:1213–1220PubMedCrossRef Hagell P, Nordling S, Reimer J, Grabowski M, Persson U (2002) Resource use and costs in a Swedish cohort of patients with Parkinson’s disease. Mov Disord 17:1213–1220PubMedCrossRef
14.
go back to reference Harrell FE (2001) Regression modeling strategies. Springer, New York Harrell FE (2001) Regression modeling strategies. Springer, New York
15.
go back to reference Hoehn MM, Yahr M (1967) Parkinsonism: onset, progression and mortality. Neurology 5:427–442 Hoehn MM, Yahr M (1967) Parkinsonism: onset, progression and mortality. Neurology 5:427–442
16.
go back to reference Litvan I, Agid Y, Calne D, Campbell G, Dubois B, Duvoisin RC, Goetz CG, Golbe LI, Grafman J, Growdon JH, Hallett M, Jankovic J, Quinn NP, Tolosa E, Zee DS (1996) Clinical research criteria for the diagnosis of progressive supranuclear palsy (Steele–Richardson–Olszewski syndrome): report of the NINDS-SPSP international workshop. Neurology 47:1–9PubMed Litvan I, Agid Y, Calne D, Campbell G, Dubois B, Duvoisin RC, Goetz CG, Golbe LI, Grafman J, Growdon JH, Hallett M, Jankovic J, Quinn NP, Tolosa E, Zee DS (1996) Clinical research criteria for the diagnosis of progressive supranuclear palsy (Steele–Richardson–Olszewski syndrome): report of the NINDS-SPSP international workshop. Neurology 47:1–9PubMed
17.
go back to reference McCrone P, Allcock LM, Burn DJ (2007) Predicting the cost of Parkinson’s disease. Mov Disord 22:804–812PubMedCrossRef McCrone P, Allcock LM, Burn DJ (2007) Predicting the cost of Parkinson’s disease. Mov Disord 22:804–812PubMedCrossRef
19.
go back to reference O’Sullivan SS, Massey LA, Williams DR, Silveira-Moriyama L, Kempster PA, Holton JL, Revesz T, Lees AJ (2008) Clinical outcomes of progressive supranuclear palsy and multiple system atrophy. Brain 131:1362–1372PubMedCrossRef O’Sullivan SS, Massey LA, Williams DR, Silveira-Moriyama L, Kempster PA, Holton JL, Revesz T, Lees AJ (2008) Clinical outcomes of progressive supranuclear palsy and multiple system atrophy. Brain 131:1362–1372PubMedCrossRef
20.
go back to reference Reese JP, Winter Y, Balzer-Geldsetzer M, Botzel K, Eggert K, Oertel WH, Dodel R, Campenhausen S (2011) Parkinson’s disease: cost-of-illness in an outpatient cohort. Gesundheitswesen 73:22–29PubMedCrossRef Reese JP, Winter Y, Balzer-Geldsetzer M, Botzel K, Eggert K, Oertel WH, Dodel R, Campenhausen S (2011) Parkinson’s disease: cost-of-illness in an outpatient cohort. Gesundheitswesen 73:22–29PubMedCrossRef
21.
go back to reference Reese JP, Winter Y, Rosa MM, Rodrigues ESAM, von Campenhausen S, Freire R, Mateus C, Balzer-Geldsetzer M, Botzel K, Oertel WH, Dodel R, Sampaio C (2011) Health-economic burden of Parkinson’s disease in Portugal: a cohort study. Revista de neurologia 52:264–274PubMed Reese JP, Winter Y, Rosa MM, Rodrigues ESAM, von Campenhausen S, Freire R, Mateus C, Balzer-Geldsetzer M, Botzel K, Oertel WH, Dodel R, Sampaio C (2011) Health-economic burden of Parkinson’s disease in Portugal: a cohort study. Revista de neurologia 52:264–274PubMed
22.
go back to reference Schepelmann K, Winter Y, Spottke AE, Claus D, Grothe C, Schroder R, Heuss D, Vielhaber S, Mylius V, Kiefer R, Schrank B, Oertel WH, Dodel R (2010) Socioeconomic burden of amyotrophic lateral sclerosis, myasthenia gravis and facioscapulohumeral muscular dystrophy. J Neurol 257:15–23PubMedCrossRef Schepelmann K, Winter Y, Spottke AE, Claus D, Grothe C, Schroder R, Heuss D, Vielhaber S, Mylius V, Kiefer R, Schrank B, Oertel WH, Dodel R (2010) Socioeconomic burden of amyotrophic lateral sclerosis, myasthenia gravis and facioscapulohumeral muscular dystrophy. J Neurol 257:15–23PubMedCrossRef
23.
go back to reference Schrag A, Ben-Shlomo Y, Quinn NP (1999) Prevalence of progressive supranuclear palsy and multiple system atrophy: a cross-sectional study. Lancet 354:1771–1775PubMedCrossRef Schrag A, Ben-Shlomo Y, Quinn NP (1999) Prevalence of progressive supranuclear palsy and multiple system atrophy: a cross-sectional study. Lancet 354:1771–1775PubMedCrossRef
26.
go back to reference The Federation of German Pharmaceutical Industry (2006) Rote Liste. Rote Liste Verlag, Weinheim The Federation of German Pharmaceutical Industry (2006) Rote Liste. Rote Liste Verlag, Weinheim
27.
go back to reference Vanacore N, Bonifati V, Fabbrini G, Colosimo C, De Michele G, Marconi R, Nicholl D, Locuratolo N, Talarico G, Romano S, Stocchi F, Bonuccelli U, De Mari M, Vieregge P, Meco G (2001) Epidemiology of multiple system atrophy. ESGAP Consortium. European Study Group on Atypical Parkinsonisms. Neurol Sci 22:97–99PubMedCrossRef Vanacore N, Bonifati V, Fabbrini G, Colosimo C, De Michele G, Marconi R, Nicholl D, Locuratolo N, Talarico G, Romano S, Stocchi F, Bonuccelli U, De Mari M, Vieregge P, Meco G (2001) Epidemiology of multiple system atrophy. ESGAP Consortium. European Study Group on Atypical Parkinsonisms. Neurol Sci 22:97–99PubMedCrossRef
28.
go back to reference von Campenhausen S, Winter Y, Gasser J, Seppi K, Reese JP, Pfeiffer KP, Geiger-Gritsch S, Botzel K, Siebert U, Oertel WH, Dodel R, Poewe W (2009) Cost of illness and health services patterns in Morbus Parkinson in Austria. Wien Klin Wochenschr 121:574–582CrossRef von Campenhausen S, Winter Y, Gasser J, Seppi K, Reese JP, Pfeiffer KP, Geiger-Gritsch S, Botzel K, Siebert U, Oertel WH, Dodel R, Poewe W (2009) Cost of illness and health services patterns in Morbus Parkinson in Austria. Wien Klin Wochenschr 121:574–582CrossRef
29.
go back to reference Vossius C, Gjerstad M, Baas H, Larsen JP (2006) Drug costs for patients with Parkinson’s disease in two different European countries. Acta Neurol Scand 113:228–232PubMedCrossRef Vossius C, Gjerstad M, Baas H, Larsen JP (2006) Drug costs for patients with Parkinson’s disease in two different European countries. Acta Neurol Scand 113:228–232PubMedCrossRef
30.
go back to reference Wenning GK, Stefanova N (2009) Recent developments in multiple system atrophy. J Neurol 256:1791–1808PubMedCrossRef Wenning GK, Stefanova N (2009) Recent developments in multiple system atrophy. J Neurol 256:1791–1808PubMedCrossRef
31.
go back to reference Wenning GK, Tison F, Seppi K, Sampaio C, Diem A, Yekhlef F, Ghorayeb I, Ory F, Galitzky M, Scaravilli T, Bozi M, Colosimo C, Gilman S, Shults CW, Quinn NP, Rascol O, Poewe W (2004) Development and validation of the Unified Multiple System Atrophy Rating Scale (UMSARS). Mov Disord 19:1391–1402PubMedCrossRef Wenning GK, Tison F, Seppi K, Sampaio C, Diem A, Yekhlef F, Ghorayeb I, Ory F, Galitzky M, Scaravilli T, Bozi M, Colosimo C, Gilman S, Shults CW, Quinn NP, Rascol O, Poewe W (2004) Development and validation of the Unified Multiple System Atrophy Rating Scale (UMSARS). Mov Disord 19:1391–1402PubMedCrossRef
32.
go back to reference Winter Y, Balzer-Geldsetzer M, Spottke A, Reese JP, Baum E, Klotsche J, Rieke J, Simonow A, Eggert K, Oertel WH, Dodel R (2010) Longitudinal study of the socioeconomic burden of Parkinson’s disease in Germany. Eur J Neurol 17:1156–1163PubMedCrossRef Winter Y, Balzer-Geldsetzer M, Spottke A, Reese JP, Baum E, Klotsche J, Rieke J, Simonow A, Eggert K, Oertel WH, Dodel R (2010) Longitudinal study of the socioeconomic burden of Parkinson’s disease in Germany. Eur J Neurol 17:1156–1163PubMedCrossRef
33.
go back to reference Winter Y, Balzer-Geldsetzer M, von Campenhausen S, Spottke A, Eggert K, Oertel WH, Dodel R (2010) Trends in resource utilization for Parkinson’s disease in Germany. J Neurol Sci 294:18–22PubMedCrossRef Winter Y, Balzer-Geldsetzer M, von Campenhausen S, Spottke A, Eggert K, Oertel WH, Dodel R (2010) Trends in resource utilization for Parkinson’s disease in Germany. J Neurol Sci 294:18–22PubMedCrossRef
34.
go back to reference Winter Y, Bezdolnyy Y, Katunina E, Avakjan G, Reese JP, Klotsche J, Oertel WH, Dodel R, Gusev E (2010) Incidence of Parkinson’s disease and atypical Parkinsonism: Russian population-based study. Mov Disord 25:349–356PubMedCrossRef Winter Y, Bezdolnyy Y, Katunina E, Avakjan G, Reese JP, Klotsche J, Oertel WH, Dodel R, Gusev E (2010) Incidence of Parkinson’s disease and atypical Parkinsonism: Russian population-based study. Mov Disord 25:349–356PubMedCrossRef
35.
go back to reference Winter Y, von Campenhausen S, Brozova H, Skoupa J, Reese JP, Botzel K, Eggert K, Oertel WH, Dodel R, Ruzicka E (2010) Costs of Parkinson’s disease in Eastern Europe: A Czech cohort study. Parkinsonism Relat Disord 16:51–56PubMedCrossRef Winter Y, von Campenhausen S, Brozova H, Skoupa J, Reese JP, Botzel K, Eggert K, Oertel WH, Dodel R, Ruzicka E (2010) Costs of Parkinson’s disease in Eastern Europe: A Czech cohort study. Parkinsonism Relat Disord 16:51–56PubMedCrossRef
36.
go back to reference Winter Y, von Campenhausen S, Popov G, Reese JP, Klotsche J, Botzel K, Gusev E, Oertel WH, Dodel R, Guekht A (2009) Costs of illness in a Russian cohort of patients with Parkinson’s disease. PharmacoEconomics 27:571–584PubMedCrossRef Winter Y, von Campenhausen S, Popov G, Reese JP, Klotsche J, Botzel K, Gusev E, Oertel WH, Dodel R, Guekht A (2009) Costs of illness in a Russian cohort of patients with Parkinson’s disease. PharmacoEconomics 27:571–584PubMedCrossRef
37.
go back to reference Winter Y, von Campenhausen S, Reese JP, Balzer-Geldsetzer M, Longo K, Spiga G, Boetzel K, Eggert K, Oertel WH, Dodel R, Barone P (2010) Costs of Parkinson’s disease and antiparkinsonian pharmacotherapy: an Italian cohort study. Neurodegener Dis 7:365–372PubMedCrossRef Winter Y, von Campenhausen S, Reese JP, Balzer-Geldsetzer M, Longo K, Spiga G, Boetzel K, Eggert K, Oertel WH, Dodel R, Barone P (2010) Costs of Parkinson’s disease and antiparkinsonian pharmacotherapy: an Italian cohort study. Neurodegener Dis 7:365–372PubMedCrossRef
Metadata
Title
Cost-of-illness in multiple system atrophy and progressive supranuclear palsy
Authors
Yaroslav Winter
Maria Stamelou
Nicole Cabanel
Friedericke Sixel-Döring
Karla Eggert
Günter U. Höglinger
Birgit Herting
Thomas Klockgether
Heinz Reichmann
Wolfgang H. Oertel
Richard Dodel
Annika E. Spottke
Publication date
01-10-2011
Publisher
Springer-Verlag
Published in
Journal of Neurology / Issue 10/2011
Print ISSN: 0340-5354
Electronic ISSN: 1432-1459
DOI
https://doi.org/10.1007/s00415-011-6028-7

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