Skip to main content
Erschienen in: The European Journal of Health Economics 1/2016

04.04.2016 | Original Paper

Social/economic costs and health-related quality of life in patients with histiocytosis in Europe

verfasst von: Georgi Iskrov, Itziar Astigarraga, Rumen Stefanov, Julio López-Bastida, Renata Linertová, Juan Oliva-Moreno, Pedro Serrano-Aguilar, Manuel Posada-de-la-Paz, Arrigo Schieppati, Domenica Taruscio, Márta Péntek, Johann Matthias Graf von der Schulenburg, Panos Kanavos, Karine Chevreul, Ulf Persson, Giovanni Fattore, BURQOL-RD Research Network

Erschienen in: The European Journal of Health Economics | Sonderheft 1/2016

Einloggen, um Zugang zu erhalten

Abstract

Objective

The aim of this study was to determine the economic burden from a societal perspective and the health-related quality of life (HRQOL) of patients with histiocytosis in Europe.

Methods

We conducted a cross-sectional study of patients with histiocytosis from France, Germany, Italy, Spain, Bulgaria, the UK, and Sweden. Data on demographic characteristics, health resource utilisation, informal care, loss of labour productivity and HRQOL were collected from the questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D) questionnaire.

Results

A total of 134 patients (35 France, 32 Germany, 30 Italy, 24 Spain, 7 Bulgaria, 4 UK and 2 Sweden) completed the questionnaire. The average annual costs ranged from € 6832 to € 33,283 between countries, the year of reference being 2012. Estimated direct healthcare costs ranged from € 1698 to € 18,213; direct nonhealthcare costs ranged from € 2936 to € 17,622 and labour productivity losses ranged from € 1 to € 8855. The mean EQ-5D score for adult histiocytosis patients was estimated at between 0.32 and 0.85, and the mean EQ-5D visual analogue scale score was estimated at between 50.00 and 66.50.

Conclusion

The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of histiocytosis patients from a broad perspective (societal costs). This type of analysis is very scarce in international literature for rare diseases in comparison with other illnesses. We conclude that histiocytosis patients incur considerable societal costs and experience substantial deterioration in HRQOL.
Anhänge
Nur mit Berechtigung zugänglich
Literatur
1.
Zurück zum Zitat Hervier, B., Haroche, J., Arnaud, L., Charlotte, F., Donadieu, J., et al.: Association of both Langerhans cell histiocytosis and Erdheim-Chester disease linked to the BRAFV600E mutation. Blood 124, 1119–1126 (2014)CrossRefPubMed Hervier, B., Haroche, J., Arnaud, L., Charlotte, F., Donadieu, J., et al.: Association of both Langerhans cell histiocytosis and Erdheim-Chester disease linked to the BRAFV600E mutation. Blood 124, 1119–1126 (2014)CrossRefPubMed
3.
Zurück zum Zitat Stålemark, H., Laurencikas, E., Karis, J., Gavhed, D., Fadeel, B., et al.: Incidence of Langerhans cell histiocytosis in children: a population-based study. Pediatr. Blood Cancer 51, 76–81 (2008)CrossRefPubMed Stålemark, H., Laurencikas, E., Karis, J., Gavhed, D., Fadeel, B., et al.: Incidence of Langerhans cell histiocytosis in children: a population-based study. Pediatr. Blood Cancer 51, 76–81 (2008)CrossRefPubMed
4.
Zurück zum Zitat Mataix, J., Betlloch, I.: Langerhans cell histiocytosis: an update. G. Ital. Dermatol. Venereol. 144, 119–134 (2009)PubMed Mataix, J., Betlloch, I.: Langerhans cell histiocytosis: an update. G. Ital. Dermatol. Venereol. 144, 119–134 (2009)PubMed
5.
Zurück zum Zitat Morimoto, A., Oh, Y., Shioda, Y., Kudo, K., Imamura, T.: Recent advances in Langerhans cell histiocytosis. Pediatr. Int. 56, 451–461 (2014)CrossRefPubMed Morimoto, A., Oh, Y., Shioda, Y., Kudo, K., Imamura, T.: Recent advances in Langerhans cell histiocytosis. Pediatr. Int. 56, 451–461 (2014)CrossRefPubMed
6.
Zurück zum Zitat Wilejto, M., Abla, O.: Langerhans cell histiocytosis and Erdheim-Chester disease. Curr. Opin. Rheumatol. 24, 90–96 (2012)CrossRefPubMed Wilejto, M., Abla, O.: Langerhans cell histiocytosis and Erdheim-Chester disease. Curr. Opin. Rheumatol. 24, 90–96 (2012)CrossRefPubMed
7.
Zurück zum Zitat DiCaprio, M.R., Roberts, T.T.: Diagnosis and Management of Langerhans Cell Histiocytosis. J. Am. Acad. Orthop. Surg. 22, 643–652 (2014)CrossRefPubMed DiCaprio, M.R., Roberts, T.T.: Diagnosis and Management of Langerhans Cell Histiocytosis. J. Am. Acad. Orthop. Surg. 22, 643–652 (2014)CrossRefPubMed
8.
Zurück zum Zitat Martin, A., Macmillan, S., Murphy, D., Carachi, R.: Langerhans cell histiocytosis: 23 years’ paediatric experience highlights severe long-term sequelae. Scott. Med. J. 59, 149–157 (2014)CrossRefPubMed Martin, A., Macmillan, S., Murphy, D., Carachi, R.: Langerhans cell histiocytosis: 23 years’ paediatric experience highlights severe long-term sequelae. Scott. Med. J. 59, 149–157 (2014)CrossRefPubMed
9.
Zurück zum Zitat Aricò, M., Girschikofsky, M., Généreau, T., Klersy, C., McClain, K., et al.: Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur. J. Cancer 39, 2341–2348 (2003)CrossRefPubMed Aricò, M., Girschikofsky, M., Généreau, T., Klersy, C., McClain, K., et al.: Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur. J. Cancer 39, 2341–2348 (2003)CrossRefPubMed
10.
Zurück zum Zitat Girschikofsky, M., Arico, M., Castillo, D., Chu, A., Doberauer, C., et al.: Management of adult patients with Langerhans cell histiocytosis: recommendations from an expert panel on behalf of Euro-Histio-Net. Orphanet. J. Rare Dis. (2013). doi:10.1186/1750-1172-8-72 PubMedPubMedCentral Girschikofsky, M., Arico, M., Castillo, D., Chu, A., Doberauer, C., et al.: Management of adult patients with Langerhans cell histiocytosis: recommendations from an expert panel on behalf of Euro-Histio-Net. Orphanet. J. Rare Dis. (2013). doi:10.​1186/​1750-1172-8-72 PubMedPubMedCentral
11.
Zurück zum Zitat Linertová, R., Serrano-Aguilar, P., Posada de la Paz, M., Hens-Pérez, M., Kanavos, P., et al.: Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study. Health Policy 108, 19–26 (2012)CrossRefPubMed Linertová, R., Serrano-Aguilar, P., Posada de la Paz, M., Hens-Pérez, M., Kanavos, P., et al.: Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study. Health Policy 108, 19–26 (2012)CrossRefPubMed
13.
Zurück zum Zitat Drummond, M.F., O’Brien, B., Stoddart, G.L., Torrance, G.W.: Methods for the economic evaluation of health care programmes, 2nd edn. Oxford University Press, Oxford (1997) Drummond, M.F., O’Brien, B., Stoddart, G.L., Torrance, G.W.: Methods for the economic evaluation of health care programmes, 2nd edn. Oxford University Press, Oxford (1997)
14.
Zurück zum Zitat McDaid, D.: Estimating the costs of informal care for people with Alzheimer’s disease: methodological and practical challenges. Int. J. Geriatr. Psychiatry 16, 400–405 (2001)CrossRefPubMed McDaid, D.: Estimating the costs of informal care for people with Alzheimer’s disease: methodological and practical challenges. Int. J. Geriatr. Psychiatry 16, 400–405 (2001)CrossRefPubMed
15.
Zurück zum Zitat Van den Berg, B., Brouwer, W., Koopmanschap, M.: Economic valuation of informal care: an overview of methods and applications. Eur. J. Health Econ 5, 36–45 (2004)CrossRefPubMed Van den Berg, B., Brouwer, W., Koopmanschap, M.: Economic valuation of informal care: an overview of methods and applications. Eur. J. Health Econ 5, 36–45 (2004)CrossRefPubMed
16.
Zurück zum Zitat Hodgson, T.A., Meiners, M.R.: Cost-of-illness methodology: a guide to assessment practices and procedures. Milbank Mem Fund Q 60, 429–491 (1982)CrossRef Hodgson, T.A., Meiners, M.R.: Cost-of-illness methodology: a guide to assessment practices and procedures. Milbank Mem Fund Q 60, 429–491 (1982)CrossRef
17.
18.
Zurück zum Zitat Mahoney, F.I., Barthel, D.W.: Functional evaluation: the Barthel Index. Md State Med 14, 61–65 (1965) Mahoney, F.I., Barthel, D.W.: Functional evaluation: the Barthel Index. Md State Med 14, 61–65 (1965)
19.
Zurück zum Zitat Shah, S., Vanclay, F., Cooper, B.: Improving the sensitivity of the Barthel Index for stroke rehabilitation. J. Clin. Epidemiol. 42, 703–709 (1989)CrossRefPubMed Shah, S., Vanclay, F., Cooper, B.: Improving the sensitivity of the Barthel Index for stroke rehabilitation. J. Clin. Epidemiol. 42, 703–709 (1989)CrossRefPubMed
20.
Zurück zum Zitat Hérbert, R., Bravo, G., Préville, M.: Reliability, validity, and reference values of the Zarit Burden Interview for assessing informal caregivers of community-dwelling older persons with dementia. Can J Aging 19, 494–507 (2000)CrossRef Hérbert, R., Bravo, G., Préville, M.: Reliability, validity, and reference values of the Zarit Burden Interview for assessing informal caregivers of community-dwelling older persons with dementia. Can J Aging 19, 494–507 (2000)CrossRef
21.
Zurück zum Zitat Imrie, J., Galani, C., Gairy, K., Lock, K., Hunsche, E.: Cost of illness associated with Niemann-Pick disease type C in the UK. J Med. Econ 12, 219–229 (2009)CrossRefPubMed Imrie, J., Galani, C., Gairy, K., Lock, K., Hunsche, E.: Cost of illness associated with Niemann-Pick disease type C in the UK. J Med. Econ 12, 219–229 (2009)CrossRefPubMed
22.
Zurück zum Zitat Klein, A.D., Alvarez, A., Zanlungo, S.: The unique case of the Niemann-Pick type C cholesterol storage disorder. Pediatr. Endocrinol. Rev. 12, 166–175 (2014)PubMed Klein, A.D., Alvarez, A., Zanlungo, S.: The unique case of the Niemann-Pick type C cholesterol storage disorder. Pediatr. Endocrinol. Rev. 12, 166–175 (2014)PubMed
23.
Zurück zum Zitat Kirchhoff, A.C., Fluchel, M.N., Wright, J., Ying, J., Sweeney, C., et al.: Risk of hospitalization for survivors of childhood and adolescent cancer. Cancer Epidemiol Biomarkers Prev 23, 1280–1289 (2014)CrossRefPubMed Kirchhoff, A.C., Fluchel, M.N., Wright, J., Ying, J., Sweeney, C., et al.: Risk of hospitalization for survivors of childhood and adolescent cancer. Cancer Epidemiol Biomarkers Prev 23, 1280–1289 (2014)CrossRefPubMed
24.
Zurück zum Zitat Audino, A.N., Yeager, N.D., Asti, L., Miao, Y., O’Brien, S.H.: Length of stay and treatment-related complications are similar in pediatric and AYA patients with bone sarcoma in United States children’s hospitals. Pediatr. Blood Cancer 60, 415–419 (2012)CrossRefPubMed Audino, A.N., Yeager, N.D., Asti, L., Miao, Y., O’Brien, S.H.: Length of stay and treatment-related complications are similar in pediatric and AYA patients with bone sarcoma in United States children’s hospitals. Pediatr. Blood Cancer 60, 415–419 (2012)CrossRefPubMed
25.
Zurück zum Zitat Warner, E.L., Kirchhoff, A.C., Nam, G.E., Fluchel, M.: Financial Burden of Pediatric Cancer for Patients and Their Families. J Oncol Pract (2014) Warner, E.L., Kirchhoff, A.C., Nam, G.E., Fluchel, M.: Financial Burden of Pediatric Cancer for Patients and Their Families. J Oncol Pract (2014)
26.
Zurück zum Zitat Wakefield, C.E., McLoone, J.K., Evans, N.T., Ellis, S.J., Cohn, R.J.: It’s more than dollars and cents: the impact of childhood cancer on parents’ occupational and financial health. J. Psychosoc. Oncol. 32, 602–621 (2014)CrossRefPubMed Wakefield, C.E., McLoone, J.K., Evans, N.T., Ellis, S.J., Cohn, R.J.: It’s more than dollars and cents: the impact of childhood cancer on parents’ occupational and financial health. J. Psychosoc. Oncol. 32, 602–621 (2014)CrossRefPubMed
27.
Zurück zum Zitat Fluchel, M.N., Kirchhoff, A.C., Bodson, J., Sweeney, C., Edwards, S.L.: Geography and the burden of care in pediatric cancers. Pediatr. Blood Cancer 61, 1918–1924 (2014)CrossRefPubMedPubMedCentral Fluchel, M.N., Kirchhoff, A.C., Bodson, J., Sweeney, C., Edwards, S.L.: Geography and the burden of care in pediatric cancers. Pediatr. Blood Cancer 61, 1918–1924 (2014)CrossRefPubMedPubMedCentral
28.
Zurück zum Zitat Kirchhoff, A.C., Lyles, C.R., Fluchel, M., Wright, J., Leisenring, W.: Limitations in health care access and utilization among long-term survivors of adolescent and young adult cancer. Cancer 118, 5964–5972 (2012)CrossRefPubMed Kirchhoff, A.C., Lyles, C.R., Fluchel, M., Wright, J., Leisenring, W.: Limitations in health care access and utilization among long-term survivors of adolescent and young adult cancer. Cancer 118, 5964–5972 (2012)CrossRefPubMed
29.
Zurück zum Zitat Dussel, V., Bona, K., Heath, J.A., Hilden, J.M., Weeks, J.C., et al.: Unmeasured costs of a child’s death: perceived financial burden, work disruptions, and economic coping strategies used by American and Australian families who lost children to cancer. J. Clin. Oncol. 29, 1007–1013 (2011)CrossRefPubMed Dussel, V., Bona, K., Heath, J.A., Hilden, J.M., Weeks, J.C., et al.: Unmeasured costs of a child’s death: perceived financial burden, work disruptions, and economic coping strategies used by American and Australian families who lost children to cancer. J. Clin. Oncol. 29, 1007–1013 (2011)CrossRefPubMed
30.
Zurück zum Zitat Vrijmoet-Wiersma, C.M., Kooloos, V.M., Koopman, H.M., Kolk, A.M., van der Laan, I., et al.: Health-related quality of life, cognitive functioning and behaviour problems in children with Langerhans cell histiocytosis. Pediatr. Blood Cancer 52, 116–122 (2009)CrossRefPubMed Vrijmoet-Wiersma, C.M., Kooloos, V.M., Koopman, H.M., Kolk, A.M., van der Laan, I., et al.: Health-related quality of life, cognitive functioning and behaviour problems in children with Langerhans cell histiocytosis. Pediatr. Blood Cancer 52, 116–122 (2009)CrossRefPubMed
31.
Zurück zum Zitat Nanduri, V.R., Pritchard, J., Levitt, G., Glaser, A.W.: Long term morbidity and health related quality of life after multi-system Langerhans cell histiocytosis. Eur. J. Cancer 42, 2563–2569 (2006)CrossRefPubMed Nanduri, V.R., Pritchard, J., Levitt, G., Glaser, A.W.: Long term morbidity and health related quality of life after multi-system Langerhans cell histiocytosis. Eur. J. Cancer 42, 2563–2569 (2006)CrossRefPubMed
32.
Zurück zum Zitat Lau, L.M., Stuurman, K., Weitzman, S.: Skeletal Langerhans cell histiocytosis in children: permanent consequences and health-related quality of life in long-term survivors. Pediatr. Blood Cancer 50, 607–612 (2008)CrossRefPubMed Lau, L.M., Stuurman, K., Weitzman, S.: Skeletal Langerhans cell histiocytosis in children: permanent consequences and health-related quality of life in long-term survivors. Pediatr. Blood Cancer 50, 607–612 (2008)CrossRefPubMed
33.
Zurück zum Zitat Laurencikas, E., Gavhed, D., Stålemark, H., van’t Hooft, I., Prayer, D., et al.: Incidence and pattern of radiological central nervous system Langerhans cell histiocytosis in children: a population based study. Pediatr. Blood Cancer 56, 250–257 (2011)CrossRefPubMed Laurencikas, E., Gavhed, D., Stålemark, H., van’t Hooft, I., Prayer, D., et al.: Incidence and pattern of radiological central nervous system Langerhans cell histiocytosis in children: a population based study. Pediatr. Blood Cancer 56, 250–257 (2011)CrossRefPubMed
34.
Zurück zum Zitat Vassallo, R., Ryu, J.H., Schroeder, D.R., Decker, P.A., Limper, A.H.: Clinical outcomes of pulmonary Langerhans’-cell histiocytosis in adults. N. Engl. J. Med. 346, 484–490 (2002)CrossRefPubMed Vassallo, R., Ryu, J.H., Schroeder, D.R., Decker, P.A., Limper, A.H.: Clinical outcomes of pulmonary Langerhans’-cell histiocytosis in adults. N. Engl. J. Med. 346, 484–490 (2002)CrossRefPubMed
35.
Zurück zum Zitat Koopman, H.M., Koetsier, J.A., Taminiau, A.H., Hijnen, K.E., Bresters, D., et al.: Health-related quality of life and coping strategies of children after treatment of a malignant bone tumor: a 5-year follow-up study. Pediatr. Blood Cancer 45, 694–699 (2005)CrossRefPubMed Koopman, H.M., Koetsier, J.A., Taminiau, A.H., Hijnen, K.E., Bresters, D., et al.: Health-related quality of life and coping strategies of children after treatment of a malignant bone tumor: a 5-year follow-up study. Pediatr. Blood Cancer 45, 694–699 (2005)CrossRefPubMed
36.
Zurück zum Zitat Simms, S., Warner, N.J.: A framework for understanding and responding to the psychosocial needs of children with Langerhans cell histiocytosis and their families. Hematol. Oncol. Clin. North Am. 12, 359–367 (1998)CrossRefPubMed Simms, S., Warner, N.J.: A framework for understanding and responding to the psychosocial needs of children with Langerhans cell histiocytosis and their families. Hematol. Oncol. Clin. North Am. 12, 359–367 (1998)CrossRefPubMed
37.
Zurück zum Zitat Mason, R.H., Foley, N.M., Branley, H.M., Adamali, H.I., Hetzel, M., et al.: Pulmonary Langerhans cell histiocytosis (PLCH): a new UK register. Thorax 69, 766–767 (2014)CrossRefPubMed Mason, R.H., Foley, N.M., Branley, H.M., Adamali, H.I., Hetzel, M., et al.: Pulmonary Langerhans cell histiocytosis (PLCH): a new UK register. Thorax 69, 766–767 (2014)CrossRefPubMed
38.
Zurück zum Zitat Taruscio, D., Gentile, A.E., Evangelista, T., Frazzica, R.G., Bushby, K., et al.: Centres of Expertise and European Reference Networks: key issues in the field of rare diseases. The EUCERD Recommendations. Blood Transfus Suppl 3, 621–625 (2014) Taruscio, D., Gentile, A.E., Evangelista, T., Frazzica, R.G., Bushby, K., et al.: Centres of Expertise and European Reference Networks: key issues in the field of rare diseases. The EUCERD Recommendations. Blood Transfus Suppl 3, 621–625 (2014)
Metadaten
Titel
Social/economic costs and health-related quality of life in patients with histiocytosis in Europe
verfasst von
Georgi Iskrov
Itziar Astigarraga
Rumen Stefanov
Julio López-Bastida
Renata Linertová
Juan Oliva-Moreno
Pedro Serrano-Aguilar
Manuel Posada-de-la-Paz
Arrigo Schieppati
Domenica Taruscio
Márta Péntek
Johann Matthias Graf von der Schulenburg
Panos Kanavos
Karine Chevreul
Ulf Persson
Giovanni Fattore
BURQOL-RD Research Network
Publikationsdatum
04.04.2016
Verlag
Springer Berlin Heidelberg
Erschienen in
The European Journal of Health Economics / Ausgabe Sonderheft 1/2016
Print ISSN: 1618-7598
Elektronische ISSN: 1618-7601
DOI
https://doi.org/10.1007/s10198-016-0790-5

Weitere Artikel der Sonderheft 1/2016

The European Journal of Health Economics 1/2016 Zur Ausgabe