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Erschienen in: The European Journal of Health Economics 7/2015

01.09.2015 | Original Paper

Cost of care for cystic fibrosis: an investigation of cost determinants using national registry data

verfasst von: Yuanyuan Gu, Sonia García-Pérez, John Massie, Kees van Gool

Erschienen in: The European Journal of Health Economics | Ausgabe 7/2015

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Abstract

Cystic fibrosis (CF) is a progressive disease with treatments intensifying as patients get older and severity worsens. To inform policy makers about the cost burden in CF, it is crucial to understand what factors influence the costs and how they affect the costs. Based on 1,060 observations (from 731 patients) obtained from the Australian Data Registry, individual annual health care costs were calculated and a regression analysis was carried out to examine the impact of multiple variables on the costs. A method of retransformation and a hypothetical patient were used for cost analysis. We show that an additional one unit improvement of FEV1pp (i.e., forced expiratory volume in 1 s as a percentage of predicted volume) reduces the costs by 1.4 %, or for a hypothetical patient whose FEV1pp is 73 the cost reduction is A$252. The presence of chronic infections increases the costs by 69.9–163.5 % (A$12,852–A$30,047 for the hypothetical patient) depending on the type of infection. The type of CF genetic mutation and the patient’s age both have significant effects on the costs. In particular, being homozygous for p.F508del increases the costs by 26.8 % compared to all the other gene mutations. We conclude that bacterial infections have a very strong influence on the costs, so reducing both the infection rates and the severity of the condition may lead to substantial cost savings. We also suggest that the patient’s genetic profile should be considered as an important cost determinant.
Fußnoten
1
These studies were identified through a snowballing approach which started from a list of papers identified in the literature reviews conducted by Krauth et al. [19], van Gool et al. [34], and Heimeshoff et al. [12]. Several rounds of searching were conducted until no extra studies could be found.
 
2
The Shapiro–Wilk test tends to reject normality when the sample size is large and our study is based on a relatively large sample (n = 1,060). Visualisation of the residuals did not show significant deviation from normality.
 
3
The hypothetical patient was created based on the sample means of independent variables.
 
Literatur
1.
Zurück zum Zitat Cystic fibrosis in Australia: 12th annual report from the Australian cystic fibrosis data registry, p. 2009. Australia, Cystic Fibrosis Australia (2009) Cystic fibrosis in Australia: 12th annual report from the Australian cystic fibrosis data registry, p. 2009. Australia, Cystic Fibrosis Australia (2009)
2.
Zurück zum Zitat AIHW. Health expenditure for arthritis and musculoskeletal conditions in Australia, 2000–01. Canberra: Australian Institute of Health and Welfare (2006) AIHW. Health expenditure for arthritis and musculoskeletal conditions in Australia, 2000–01. Canberra: Australian Institute of Health and Welfare (2006)
3.
Zurück zum Zitat Baumann, U., Stocklossa, C., Greiner, W., von der Schulenburg, J.M., von der Hardt, H.: Cost of care and clinical condition in paediatric cystic fibrosis patients. J Cyst Fibros 2, 84–90 (2003)CrossRefPubMed Baumann, U., Stocklossa, C., Greiner, W., von der Schulenburg, J.M., von der Hardt, H.: Cost of care and clinical condition in paediatric cystic fibrosis patients. J Cyst Fibros 2, 84–90 (2003)CrossRefPubMed
4.
Zurück zum Zitat Colagiuri, S., Colagiuri, R., Grainger, D., Graham-Clarke, P., Davey, P., Fitzgerald, P., Conway, B.: Diabcost Australia—assessing the burden of type 2 diabetes in Australia. Diabetologia 45, A300–A300 (2002) Colagiuri, S., Colagiuri, R., Grainger, D., Graham-Clarke, P., Davey, P., Fitzgerald, P., Conway, B.: Diabcost Australia—assessing the burden of type 2 diabetes in Australia. Diabetologia 45, A300–A300 (2002)
5.
Zurück zum Zitat Corey, M., Farewell, V.: Determinants of mortality from cystic fibrosis in Canada, 1970–1989. Am J Epidemiol 143, 1007–1017 (1996)CrossRefPubMed Corey, M., Farewell, V.: Determinants of mortality from cystic fibrosis in Canada, 1970–1989. Am J Epidemiol 143, 1007–1017 (1996)CrossRefPubMed
6.
Zurück zum Zitat Dasenbrook, E.C., Checkley, W., Merlo, C.A., Konstan, M.W., Lechtzin, N., Boyle, M.P.: Association between respiratory tract methicillin-resistant Staphylococcus aureus and survival in cystic fibrosis. JAMA 303, 2386–2392 (2010)CrossRefPubMed Dasenbrook, E.C., Checkley, W., Merlo, C.A., Konstan, M.W., Lechtzin, N., Boyle, M.P.: Association between respiratory tract methicillin-resistant Staphylococcus aureus and survival in cystic fibrosis. JAMA 303, 2386–2392 (2010)CrossRefPubMed
7.
Zurück zum Zitat Dewitt, E.M., Grussemeyer, C.A., Friedman, J.Y., Dinan, M.A., Lin, L., Schulman, K.A., Reed, S.D.: Resource use, costs, and utility estimates for patients with cystic fibrosis with mild impairment in lung function: analysis of data collected alongside a 48-week multicenter clinical trial. Value in health 15, 277–283 (2012)CrossRefPubMed Dewitt, E.M., Grussemeyer, C.A., Friedman, J.Y., Dinan, M.A., Lin, L., Schulman, K.A., Reed, S.D.: Resource use, costs, and utility estimates for patients with cystic fibrosis with mild impairment in lung function: analysis of data collected alongside a 48-week multicenter clinical trial. Value in health 15, 277–283 (2012)CrossRefPubMed
8.
Zurück zum Zitat Dodge, J.A., Morison, S., Lewis, P.A., Coles, E.C., Geddes, D., Russell, G., Littlewood, J.M., Scott, M.T.: Incidence, population, and survival of cystic fibrosis in the UK, 1968–95.UCFSM Committee. Arch Dis Child 77, 493–496 (1997)PubMedCentralCrossRefPubMed Dodge, J.A., Morison, S., Lewis, P.A., Coles, E.C., Geddes, D., Russell, G., Littlewood, J.M., Scott, M.T.: Incidence, population, and survival of cystic fibrosis in the UK, 1968–95.UCFSM Committee. Arch Dis Child 77, 493–496 (1997)PubMedCentralCrossRefPubMed
9.
Zurück zum Zitat Duan, N.: Smearing estimate—a nonparametric retransformation method. J Am Stat Assoc 78, 605–610 (1983)CrossRef Duan, N.: Smearing estimate—a nonparametric retransformation method. J Am Stat Assoc 78, 605–610 (1983)CrossRef
10.
Zurück zum Zitat Eidt-Koch, D., Wagner, T.O., Mittendorf, T., Graf von der Schulenburg, J.M.: Outpatient medication costs of patients with cystic fibrosis in Germany. Appl Health Econ Health Policy 8, 111–118 (2010)CrossRefPubMed Eidt-Koch, D., Wagner, T.O., Mittendorf, T., Graf von der Schulenburg, J.M.: Outpatient medication costs of patients with cystic fibrosis in Germany. Appl Health Econ Health Policy 8, 111–118 (2010)CrossRefPubMed
11.
Zurück zum Zitat Frangolias, D.D., Mahenthiralingam, E., Rae, S., Raboud, J.M., Davidson, A.G.F., Wittmann, R., Wilcox, P.G.: Burkholderia cepacia in cystic fibrosis—variable disease course. Am J Respir Crit Care Med 160, 1572–1577 (1999)CrossRefPubMed Frangolias, D.D., Mahenthiralingam, E., Rae, S., Raboud, J.M., Davidson, A.G.F., Wittmann, R., Wilcox, P.G.: Burkholderia cepacia in cystic fibrosis—variable disease course. Am J Respir Crit Care Med 160, 1572–1577 (1999)CrossRefPubMed
12.
Zurück zum Zitat Heimeshoff, M., Hollmeyer, H., Schreyogg, J., Tiemann, O., Staab, D.: Cost of illness of cystic fibrosis in Germany: results from a large cystic fibrosis centre. Pharmacoeconomics 30, 763–777 (2012)CrossRefPubMed Heimeshoff, M., Hollmeyer, H., Schreyogg, J., Tiemann, O., Staab, D.: Cost of illness of cystic fibrosis in Germany: results from a large cystic fibrosis centre. Pharmacoeconomics 30, 763–777 (2012)CrossRefPubMed
13.
Zurück zum Zitat Jacques, I., Derelle, J., Weber, M., Vidailhet, M.: Pulmonary evolution of cystic fibrosis patients colonized by Pseudomonas aeruginosa and/or Burkholderia cepacia. Eur J Pediatr 157, 427–431 (1998)CrossRefPubMed Jacques, I., Derelle, J., Weber, M., Vidailhet, M.: Pulmonary evolution of cystic fibrosis patients colonized by Pseudomonas aeruginosa and/or Burkholderia cepacia. Eur J Pediatr 157, 427–431 (1998)CrossRefPubMed
14.
Zurück zum Zitat Johansen, H.K., Nir, M., Hoiby, N., Koch, C., Schwartz, M.: Severity of cystic-fibrosis in patients homozygous and heterozygous for delta-f508 mutation. Lancet 337, 631–634 (1991)CrossRefPubMed Johansen, H.K., Nir, M., Hoiby, N., Koch, C., Schwartz, M.: Severity of cystic-fibrosis in patients homozygous and heterozygous for delta-f508 mutation. Lancet 337, 631–634 (1991)CrossRefPubMed
15.
Zurück zum Zitat Johnson, J.A., Connolly, M.A., Jacobs, P., Montgomery, M., Brown, N.E., Zuberbuhler, P.: Cost of care for individuals with cystic fibrosis: a regression approach to determining the impact of recombinant human DNase. Pharmacotherapy 19, 1159–1166 (1999)CrossRefPubMed Johnson, J.A., Connolly, M.A., Jacobs, P., Montgomery, M., Brown, N.E., Zuberbuhler, P.: Cost of care for individuals with cystic fibrosis: a regression approach to determining the impact of recombinant human DNase. Pharmacotherapy 19, 1159–1166 (1999)CrossRefPubMed
16.
Zurück zum Zitat Jones, A.M.: Models for health care. In: Hendry, D., Clements, M. (eds.) Oxford handbook of economic forecasting, pp. 625–654. Oxford University Press, New York (2011) Jones, A.M.: Models for health care. In: Hendry, D., Clements, M. (eds.) Oxford handbook of economic forecasting, pp. 625–654. Oxford University Press, New York (2011)
17.
Zurück zum Zitat Kerem, E., Corey, M., Gold, R., Levison, H.: Pulmonary-function and clinical course in patients with cystic-fibrosis after pulmonary colonization with Pseudomonas aeruginosa. J Pediatr. 116, 714–719 (1990)CrossRefPubMed Kerem, E., Corey, M., Gold, R., Levison, H.: Pulmonary-function and clinical course in patients with cystic-fibrosis after pulmonary colonization with Pseudomonas aeruginosa. J Pediatr. 116, 714–719 (1990)CrossRefPubMed
18.
Zurück zum Zitat Kerem, E., Reisman, J., Corey, M., Canny, G.J., Levison, H.: Prediction of mortality in patients with cystic-fibrosis. N Engl J Med 326, 1187–1191 (1992)CrossRefPubMed Kerem, E., Reisman, J., Corey, M., Canny, G.J., Levison, H.: Prediction of mortality in patients with cystic-fibrosis. N Engl J Med 326, 1187–1191 (1992)CrossRefPubMed
19.
Zurück zum Zitat Krauth, C., Jalilvand, N., Welte, T., Busse, R.: Cystic fibrosis: cost of illness and considerations for the economic evaluation of potential therapies. Pharmacoeconomics 21, 1001–1024 (2003)CrossRefPubMed Krauth, C., Jalilvand, N., Welte, T., Busse, R.: Cystic fibrosis: cost of illness and considerations for the economic evaluation of potential therapies. Pharmacoeconomics 21, 1001–1024 (2003)CrossRefPubMed
20.
Zurück zum Zitat Lieu, T.A., Ray, G.T., Farmer, G., Shay, G.F.: The cost of medical care for patients with cystic fibrosis in a health maintenance organization. Pediatrics 103, art. no.e72 (1999) Lieu, T.A., Ray, G.T., Farmer, G., Shay, G.F.: The cost of medical care for patients with cystic fibrosis in a health maintenance organization. Pediatrics 103, art. no.e72 (1999)
21.
Zurück zum Zitat Liou, T.G., Adler, F.R., FitzSimmons, S.C., Cahill, B.C., Hibbs, J.R., Marshall, B.C.: Predictive 5-year survivorship model of cystic fibrosis. Am J Epidemiol 153, 345–352 (2001)PubMedCentralCrossRefPubMed Liou, T.G., Adler, F.R., FitzSimmons, S.C., Cahill, B.C., Hibbs, J.R., Marshall, B.C.: Predictive 5-year survivorship model of cystic fibrosis. Am J Epidemiol 153, 345–352 (2001)PubMedCentralCrossRefPubMed
22.
Zurück zum Zitat Manning, W.G., Basu, A., Mullahy, J.: Generalized modeling approaches to risk adjustment of skewed outcomes data. J Health Econ 24, 465–488 (2005)CrossRefPubMed Manning, W.G., Basu, A., Mullahy, J.: Generalized modeling approaches to risk adjustment of skewed outcomes data. J Health Econ 24, 465–488 (2005)CrossRefPubMed
23.
Zurück zum Zitat Massie, R.J., Olsen, M., Glazner, J., Robertson, C.F., Francis, I.: Newborn screening for cystic fibrosis in victoria: 10 years’ experience (1989-1998). Med J Aust 172, 584–587 (2000)PubMed Massie, R.J., Olsen, M., Glazner, J., Robertson, C.F., Francis, I.: Newborn screening for cystic fibrosis in victoria: 10 years’ experience (1989-1998). Med J Aust 172, 584–587 (2000)PubMed
24.
Zurück zum Zitat Mayer-Hamblett, N., Rosenfeld, M., Emerson, J., Goss, C.H., Aitken, M.L.: Developing cystic fibrosis lung transplant referral criteria using predictors of 2-year mortality. Am J Respir Crit Care Med 166, 1550–1555 (2002)CrossRefPubMed Mayer-Hamblett, N., Rosenfeld, M., Emerson, J., Goss, C.H., Aitken, M.L.: Developing cystic fibrosis lung transplant referral criteria using predictors of 2-year mortality. Am J Respir Crit Care Med 166, 1550–1555 (2002)CrossRefPubMed
25.
Zurück zum Zitat O’Brien, R.M.: A caution regarding rules of thumb for variance inflation factors. Qual Quant 41, 673–690 (2007)CrossRef O’Brien, R.M.: A caution regarding rules of thumb for variance inflation factors. Qual Quant 41, 673–690 (2007)CrossRef
26.
Zurück zum Zitat Ouyang, L., Grosse, S.D., Amendah, D.D., Schechter, M.S.: Healthcare expenditures for privately insured people with cystic fibrosis. Pediatr Pulmonol 44, 989–996 (2009)CrossRefPubMed Ouyang, L., Grosse, S.D., Amendah, D.D., Schechter, M.S.: Healthcare expenditures for privately insured people with cystic fibrosis. Pediatr Pulmonol 44, 989–996 (2009)CrossRefPubMed
27.
Zurück zum Zitat Pamukcu, A., Bush, A., Buchdahl, R.: Effects of Pseudomonas aeruginosa colonization on lung-function and anthropometric variables in children with cystic fibrosis. Pediatr Pulonol 19, 10–15 (1995)CrossRef Pamukcu, A., Bush, A., Buchdahl, R.: Effects of Pseudomonas aeruginosa colonization on lung-function and anthropometric variables in children with cystic fibrosis. Pediatr Pulonol 19, 10–15 (1995)CrossRef
28.
Zurück zum Zitat PBAC. Guidelines for preparing submissions to the Pharmaceutical Benefits Advisory Committee Canberra. Department of Health and Ageing (2008) PBAC. Guidelines for preparing submissions to the Pharmaceutical Benefits Advisory Committee Canberra. Department of Health and Ageing (2008)
29.
Zurück zum Zitat Ramsey, B.W., Astley, S.J., Aitken, M.L., Burke, W., Colin, A.A., Dorkin, H.L., Eisenberg, J.D., Gibson, R.L., Harwood, I.R., Schidlow, D.V., Wilmott, R.W., Wohl, M.E., Meyerson, L.J., Shak, S., Fuchs, H., Smith, A.L.: Efficacy and safety of short-term administration of aerosolized recombinant human deoxyribonuclease in patients with cystic fibrosis. Am Rev Respir Dis 148, 145–151 (1993)CrossRefPubMed Ramsey, B.W., Astley, S.J., Aitken, M.L., Burke, W., Colin, A.A., Dorkin, H.L., Eisenberg, J.D., Gibson, R.L., Harwood, I.R., Schidlow, D.V., Wilmott, R.W., Wohl, M.E., Meyerson, L.J., Shak, S., Fuchs, H., Smith, A.L.: Efficacy and safety of short-term administration of aerosolized recombinant human deoxyribonuclease in patients with cystic fibrosis. Am Rev Respir Dis 148, 145–151 (1993)CrossRefPubMed
30.
Zurück zum Zitat Schreyogg, J., Hollmeyer, H., Bluemel, M., Staab, D., Busse, R.: Hospitalisation costs of cystic fibrosis. Pharmacoeconomics 24, 999–1009 (2006)CrossRefPubMed Schreyogg, J., Hollmeyer, H., Bluemel, M., Staab, D., Busse, R.: Hospitalisation costs of cystic fibrosis. Pharmacoeconomics 24, 999–1009 (2006)CrossRefPubMed
31.
Zurück zum Zitat Serginson, J.G., Yang, I.A., Armstrong, J.G., Cooper, D.M., Matthiesson, A.M., Morrison, S.C., Gair, J.M., Cooper, B., Zimmerman, P.V.: Variability in the rate of prescription and cost of domiciliary oxygen therapy in Australia. Med J Aust 191, 549–553 (2009)PubMed Serginson, J.G., Yang, I.A., Armstrong, J.G., Cooper, D.M., Matthiesson, A.M., Morrison, S.C., Gair, J.M., Cooper, B., Zimmerman, P.V.: Variability in the rate of prescription and cost of domiciliary oxygen therapy in Australia. Med J Aust 191, 549–553 (2009)PubMed
32.
Zurück zum Zitat Singh, S.J., Sodergren, S.C., Hyland, M.E., Williams, J., Morgan, M.D.: A comparison of three disease-specific and two generic health-status measures to evaluate the outcome of pulmonary rehabilitation in COPD. Respir Med 95, 71–77 (2001)CrossRefPubMed Singh, S.J., Sodergren, S.C., Hyland, M.E., Williams, J., Morgan, M.D.: A comparison of three disease-specific and two generic health-status measures to evaluate the outcome of pulmonary rehabilitation in COPD. Respir Med 95, 71–77 (2001)CrossRefPubMed
33.
Zurück zum Zitat Stallings, V.A., Stark, L.J., Robinson, K.A., Feranchak, A.P., Quinton, H., Group, AHW: Evidence-based practice recommendations for nutrition-related management of children and adults with cystic fibrosis and pancreatic insufficiency: results of a systematic review. J Am Diet Assoc 108, 832–839 (2008)CrossRefPubMed Stallings, V.A., Stark, L.J., Robinson, K.A., Feranchak, A.P., Quinton, H., Group, AHW: Evidence-based practice recommendations for nutrition-related management of children and adults with cystic fibrosis and pancreatic insufficiency: results of a systematic review. J Am Diet Assoc 108, 832–839 (2008)CrossRefPubMed
34.
Zurück zum Zitat van Gool, K., Norman, R., Delatycki, M.B., Hall, J., Massie, J.: Understanding the costs of care for cystic fibrosis: an analysis by age and health state. Value in health 16, 345–355 (2013)CrossRefPubMed van Gool, K., Norman, R., Delatycki, M.B., Hall, J., Massie, J.: Understanding the costs of care for cystic fibrosis: an analysis by age and health state. Value in health 16, 345–355 (2013)CrossRefPubMed
35.
Zurück zum Zitat Welsh, M., Ramsey, B., Accurso, F., Cutting, G.: Cystic fibrosis. In: Scriver, C., Beaudet, A., Sly, W., Valle, D. (eds.) The metabolic and molecular bases of inherited disease, pp. 5121–5188. McGraw-Hill, New York (2001) Welsh, M., Ramsey, B., Accurso, F., Cutting, G.: Cystic fibrosis. In: Scriver, C., Beaudet, A., Sly, W., Valle, D. (eds.) The metabolic and molecular bases of inherited disease, pp. 5121–5188. McGraw-Hill, New York (2001)
36.
Zurück zum Zitat White, H.: A heteroskedasticity-consistent covariance-matrix estimator and a direct test for heteroskedasticity. Econometrica 48, 817–838 (1980)CrossRef White, H.: A heteroskedasticity-consistent covariance-matrix estimator and a direct test for heteroskedasticity. Econometrica 48, 817–838 (1980)CrossRef
Metadaten
Titel
Cost of care for cystic fibrosis: an investigation of cost determinants using national registry data
verfasst von
Yuanyuan Gu
Sonia García-Pérez
John Massie
Kees van Gool
Publikationsdatum
01.09.2015
Verlag
Springer Berlin Heidelberg
Erschienen in
The European Journal of Health Economics / Ausgabe 7/2015
Print ISSN: 1618-7598
Elektronische ISSN: 1618-7601
DOI
https://doi.org/10.1007/s10198-014-0621-5

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