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The economic burden of amyotrophic lateral sclerosis: a systematic review

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Abstract

Objectives

This systematic review aimed to comprehensively collect and summarise the current body of knowledge regarding the cost-of-illness of amyotrophic lateral sclerosis, to identify cost-driving factors of the disease and to consider the development of costs over the course of disease. Further, the review sought to assess the methodological quality of the selected studies.

Methods

A systematic review was performed using the databases MEDLINE, Embase, Cochrane Library and PsycINFO. Studies examining the economic burden of amyotrophic lateral sclerosis on a patient or national level written in English or German published from the year 2001 onwards were included. Additional searches were conducted. Study characteristics and results were extracted and compared.

Results

In summary, 20 studies were included in this review. Most studies investigated costs per patient, amounting to total costs between €9741€ to €114,605. Six studies confirmed a rise in costs with disease progression, peaking close to the death of a patient. National costs for amyotrophic lateral sclerosis varied between €149 million and €1329 million.

Conclusion

Most of these studies suggest the economic burden of amyotrophic lateral sclerosis to be considerable. However, further research is needed to establish a cost-effective health policy in consideration of disease severities.

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References

  1. Schlaile, M.P., Knausberg, T., Mueller, M., Zeman, J.: Viral ice buckets: a memetic perspective on the ALS Ice Bucket Challenge’s diffusion. Cogn. Syst. Res. 52, 947–969 (2018)

    Article  Google Scholar 

  2. Brown, R.H., Al-Chalabi, A.: Amyotrophic lateral sclerosis. N Engl J Med 377(2), 162–172 (2017). https://doi.org/10.1056/NEJMra1603471

    Article  CAS  PubMed  Google Scholar 

  3. Gastl, R., Ludolph, A.C.: Amyotrophic lateral sclerosis. Nervenarzt 78(12), 1449–1457 (2007). https://doi.org/10.1007/s00115-007-2354-5. (quiz 1458-1449)

    Article  CAS  PubMed  Google Scholar 

  4. Pupillo, E., Messina, P., Logroscino, G., Beghi, E., S Group: Long-term survival in amyotrophic lateral sclerosis: a population-based study. Ann Neurol 75(2), 287–297 (2014). https://doi.org/10.1002/ana.24096

    Article  PubMed  Google Scholar 

  5. Beghi, E., Logroscino, G., Chio, A., Hardiman, O., Mitchell, D., Swingler, R., Traynor, B.J.: The epidemiology of ALS and the role of population-based registries. Biochim Biophys Acta 1762(11–12), 1150–1157 (2006). https://doi.org/10.1016/j.bbadis.2006.09.008. (Epub 2006 Sep 29)

    Article  CAS  PubMed  Google Scholar 

  6. Logroscino, G., Traynor, B.J., Hardiman, O., Chio, A., Couratier, P., Mitchell, J.D., Swingler, R.J., Beghi, E.: Eurals: descriptive epidemiology of amyotrophic lateral sclerosis: new evidence and unsolved issues. J Neurol Neurosurg Psychiatry 79(1), 6–11 (2008). https://doi.org/10.1136/jnnp.2006.104828

    Article  CAS  PubMed  Google Scholar 

  7. Logroscino, G., Traynor, B.J., Hardiman, O., Chio, A., Mitchell, D., Swingler, R.J., Millul, A., Benn, E., Beghi, E.: Eurals: Incidence of amyotrophic lateral sclerosis in Europe. J Neurol Neurosurg Psychiatry 81(4), 385–390 (2010). https://doi.org/10.1136/jnnp.2009.183525

    Article  PubMed  Google Scholar 

  8. Chancellor, A.M., Warlow, C.P.: Adult onset motor neuron disease: worldwide mortality, incidence and distribution since 1950. J Neurol Neurosurg Psychiatry 55(12), 1106–1115 (1992). https://doi.org/10.1136/jnnp.55.12.1106

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  9. Ludolph, A. (ed.): Motoneuronenerkrankungen, 3rd edn. Springer, Berlin (2011)

    Google Scholar 

  10. Chio, A., Logroscino, G., Traynor, B.J., Collins, J., Simeone, J.C., Goldstein, L.A., White, L.A.: Global epidemiology of amyotrophic lateral sclerosis: a systematic review of the published literature. Neuroepidemiology 41(2), 118–130 (2013). https://doi.org/10.1159/000351153. (Epub 2013 Jul 11)

    Article  CAS  PubMed  Google Scholar 

  11. Brooks, B.R.: El Escorial World Federation of Neurology criteria for the diagnosis of amyotrophic lateral sclerosis. Subcommittee on Motor Neuron Diseases/Amyotrophic Lateral Sclerosis of the World Federation of Neurology Research Group on Neuromuscular Diseases and the El Escorial “Clinical limits of amyotrophic lateral sclerosis” workshop contributors. J Neurol Sci 124, 96–107 (1994). https://doi.org/10.1016/0022-510x(94)90191-0

    Article  PubMed  Google Scholar 

  12. Wijesekera, L.C., Leigh, P.N.: Amyotrophic lateral sclerosis. Orphanet J Rare Dis 4, 3 (2009). https://doi.org/10.1186/1750-1172-4-3

    Article  PubMed  PubMed Central  Google Scholar 

  13. Miller, R.G., Mitchell, J.D., Moore, D.H.: Riluzole for amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND). Cochrane Database Syst. Rev. (3), CD001447 (2012). https://doi.org/10.1002/14651858.CD001447.pub3

  14. Al-Chalabi, A., Andersen, P.M., Chandran, S., Chio, A., Corcia, P., Couratier, P., Danielsson, O., de Carvalho, M., Desnuelle, C., Grehl, T., Grosskreutz, J., Holmoy, T., Ingre, C., Karlsborg, M., Kleveland, G., Koch, J.C., Koritnik, B., KuzmaKozakiewicz, M., Laaksovirta, H., Ludolph, A., McDermott, C., Meyer, T., Mitre Ropero, B., Mora Pardina, J., Nygren, I., Petri, S., Povedano Panades, M., Salachas, F., Shaw, P., Silani, V., Staaf, G., Svenstrup, K., Talbot, K., Tysnes, O.B., Van Damme, P., van der Kooi, A., Weber, M., Weydt, P., Wolf, J., Hardiman, O., van den Berg, L.H.: July 2017 ENCALS statement on edaravone. Amyotroph Lateral Scler Frontotemporal Degener 18(7–8), 471–474 (2017). https://doi.org/10.1080/21678421.2017.1369125. (Epub 2017 Oct 4)

    Article  PubMed  Google Scholar 

  15. Liu, S.S., Rawicz, A., Rezael, S., Ma, T., Zhang, C., Lin, K.: An eye-Gaze tracking and human computer interface system for people with ALS and other locked-in diseases. J Med Biol Eng 32(2), 111–116 (2012)

    Article  Google Scholar 

  16. Ginsberg, G., Lowe, S.: Cost effectiveness of treatments for amyotrophic lateral sclerosis: a review of the literature. Pharmacoeconomics 20(6), 367–387 (2002). https://doi.org/10.2165/00019053-200220060-00002

    Article  CAS  PubMed  Google Scholar 

  17. Wynford-Thomas, R., Robertson, N.P.: The economic burden of chronic neurological disease. J Neurol 264(11), 2345–2347 (2017). https://doi.org/10.1007/s00415-017-8632-7

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  18. World Health Oranisation: Neurological disorders - public health challenges. https://www.who.int/mental_health/neurology/introduction_neuro_disorders_public_h_challenges.pdf?ua=1 (2006). Accessed June 2020

  19. Fiorentino, G., Esquinas, A.M.: Cost-effectiveness associated with amyotrophic lateral sclerosis: some questions and answers pending. Amyotroph Lateral Scler Frontotemporal Degener 19(3–4), 315–316 (2018). https://doi.org/10.1080/21678421.2017.1408126. (Epub 2017 Nov 30)

    Article  PubMed  Google Scholar 

  20. Gladman, M., Zinman, L.: The economic impact of amyotrophic lateral sclerosis: a systematic review. Expert Rev Pharmacoecon Outcomes Res 15(3), 439–450 (2015). https://doi.org/10.1586/14737167.2015.1039941. (Epub 2015 Apr 30)

    Article  PubMed  Google Scholar 

  21. Moher, D., Liberati, A., Tetzlaff, J., Altman, D.G., P Group: Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. J Clin Epidemiol 62(10), 1006–1012 (2009). https://doi.org/10.1016/j.jclinepi.2009.06.005. (Epub 2009 Jul 23)

    Article  PubMed  Google Scholar 

  22. Liberati, A., Altman, D.G., Tetzlaff, J., Mulrow, C., Gotzsche, P.C., Ioannidis, J.P., Clarke, M., Devereaux, P.J., Kleijnen, J., Moher, D.: The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration. J Clin Epidemiol 62(10), e1-34 (2009). https://doi.org/10.1016/j.jclinepi.2009.06.006. (Epub 2009 Jul 23)

    Article  Google Scholar 

  23. Higgins, J.P.T., Thomas, J., Chandler, J., Cumpston, M., Li, T., Page, M.J., Welch, V.A.: Cochrane Handbook for systematic reviews of interventions version 6.0 (updated July 2019) (2019)

  24. Kassenärztliche Bundesvereinigung: Online-Version des EBM. https://www.kbv.de/html/online-ebm.php (2020). Accessed June 2020

  25. Oecd: Purchasing Power Parties (PPP) (2020). https://data.oecd.org/conversion/purchasing-power-parities-ppp.htm. Accessed June 2020

  26. Statistisches, B.: Harmonisierter Verbraucherpreisindex (2020). https://www.destatis.de/DE/ZahlenFakten/Gesamtwirtschaft-Umwelt/Preise/Verbraucherpreisindizes/Tabellen_/HarmonisierterVerbraucherpreisindex.tml?cms_gtp=146598_slot%253D2. Accessed June 2020

  27. Drummond, M.F., Jefferson, T.O.: Guidelines for authors and peer reviewers of economic submissions to the BMJ. The BMJ economic evaluation working party. BMJ 313(7052), 275–283 (1996). https://doi.org/10.1136/bmj.313.7052.275

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  28. Einarson, T.R., Acs, A., Ludwig, C., Panton, U.H.: Economic burden of cardiovascular disease in Type 2 diabetes: a systematic review. Value. Health. 21(7), 881–890 (2018). https://doi.org/10.1016/j.jval.2017.12.019. (Epub 2018 Feb 16)

    Article  PubMed  Google Scholar 

  29. Pelone, F., Specchia, M.L., Veneziano, M.A., Capizzi, S., Bucci, S., Mancuso, A., Ricciardi, W., de Belvis, A.G.: Economic impact of childhood obesity on health systems: a systematic review. Obes. Rev. 13(5), 431–440 (2012). https://doi.org/10.1111/j.1467-789X.2011.00968.x. (Epub 2011 Dec 29)

    Article  CAS  PubMed  Google Scholar 

  30. Bahadori, K., Doyle-Waters, M.M., Marra, C., Lynd, L., Alasaly, K., Swiston, J., FitzGerald, J.M.: Economic burden of asthma: a systematic review. BMC. Pulm. Med. 9, 24 (2009). https://doi.org/10.1186/1471-2466-9-24

    Article  PubMed  PubMed Central  Google Scholar 

  31. Husereau, D., Drummond, M., Petrou, S., Carswell, C., Moher, D., Greenberg, D., Augustovski, F., Briggs, A.H., Mauskopf, J., Loder, E., Force, C.T.: Consolidated health economic evaluation reporting standards (CHEERS) statement. BJOG. 120(6), 765–770 (2013). https://doi.org/10.1111/1471-0528.12241

    Article  CAS  PubMed  Google Scholar 

  32. Evers, S., Goossens, M., de Vet, H., van Tulder, M., Ament, A.: Criteria list for assessment of methodological quality of economic evaluations: consensus on Health Economic Criteria. Int. J. Technol. Assess. Health. Care. 21(2), 240–245 (2005)

    Article  Google Scholar 

  33. Balshem, H., Helfand, M., Schunemann, H.J., Oxman, A.D., Kunz, R., Brozek, J., Vist, G.E., Falck-Ytter, Y., Meerpohl, J., Norris, S., Guyatt, G.H.: GRADE guidelines: 3. Rating the quality of evidence. J. Clin. Epidemiol. 64(4), 401–406 (2011). https://doi.org/10.1016/j.jclinepi.2010.07.015. (Epub 2011 Jan 5)

    Article  PubMed  Google Scholar 

  34. van der Steen, I., van den Berg, J.P., Buskens, E., Lindeman, E., van den Berg, L.H.: The costs of amyotrophic lateral sclerosis, according to type of care. Amyotroph. Lateral. Scler. 10(1), 27–34 (2009). https://doi.org/10.1080/17482960802103131

    Article  PubMed  Google Scholar 

  35. Athanasakis, K., Kyriopoulos, I.I., Sideris, M., Rentzos, M., Evdokimidis, J., Kyriopoulos, J.: Investigating the economic burden of ALS in Greece: a cost-of-illness approach. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 16(1–2), 63–64 (2015). https://doi.org/10.3109/21678421.2014.932384. (Epub 2014 Jul 14)

    Article  PubMed  Google Scholar 

  36. Jennum, P., Ibsen, R., Pedersen, S.W., Kjellberg, J.: Mortality, health, social and economic consequences of amyotrophic lateral sclerosis: a controlled national study. J. Neurol. 260(3), 785–793 (2013). https://doi.org/10.1007/s00415-012-6706-0. (Epub 2012 Oct 27)

    Article  PubMed  Google Scholar 

  37. Larkindale, J., Yang, W., Hogan, P.F., Simon, C.J., Zhang, Y., Jain, A., Habeeb-Louks, E.M., Kennedy, A., Cwik, V.A.: Cost of illness for neuromuscular diseases in the United States. Muscle. Nerve. 49(3), 431–438 (2014). https://doi.org/10.1002/mus.23942. (Epub 2014 Jan 28)

    Article  PubMed  Google Scholar 

  38. Muscular Dystrophy, A.: Cost of amyotrophic lateral sclerosis, muscular dystrophy, and spinal muscular atrophy in the United States (2012). https://www.mda.org/sites/default/files/Cost_Illness_Report_0.pdf

  39. Lopez-Bastida, J., Perestelo-Perez, L., Monton-Alvarez, F., Serrano-Aguilar, P., Alfonso-Sanchez, J.L.: Social economic costs and health-related quality of life in patients with amyotrophic lateral sclerosis in Spain. Amyotroph. Lateral. Scler. 10(4), 237–243 (2009). https://doi.org/10.1080/17482960802430781

    Article  PubMed  Google Scholar 

  40. Oh, J., An, J.W., Oh, S.I., Oh, K.W., Kim, J.A., Lee, J.S., Kim, S.H.: Socioeconomic costs of amyotrophic lateral sclerosis according to staging system. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 16(3–4), 202–208 (2015). https://doi.org/10.3109/21678421.2014.999791. (Epub 2015 Feb 3)

    Article  PubMed  Google Scholar 

  41. Schepelmann, K., Winter, Y., Spottke, A.E., Claus, D., Grothe, C., Schroder, R., Heuss, D., Vielhaber, S., Mylius, V., Kiefer, R., Schrank, B., Oertel, W.H., Dodel, R.: Socioeconomic burden of amyotrophic lateral sclerosis, myasthenia gravis and facioscapulohumeral muscular dystrophy. J. Neurol. 257(1), 15–23 (2010). https://doi.org/10.1007/s00415-009-5256-6. (Epub 2009 Jul 24)

    Article  PubMed  Google Scholar 

  42. Schonfelder, E., Osmanovic, A., Muschen, L.H., Petri, S., Schreiber-Katz, O.: Costs of illness in amyotrophic lateral sclerosis (ALS): a cross-sectional survey in Germany. Orphanet. J. Rare. Dis. 15(1), 149 (2020). https://doi.org/10.1186/s13023-020-01413-9

    Article  PubMed  PubMed Central  Google Scholar 

  43. Deloitte Access Economics: Economics analysis of motor neurone disease in Australia–Report for Motor Neurone Disease Australia (2015). https://www2.deloitte.com/au/en/pages/economics/articles/economic-analysis-motor-neurone-disease-australia.html

  44. Connolly, S., Heslin, C., Mays, I., Corr, B., Normand, C., Hardiman, O.: Health and social care costs of managing amyotrophic lateral sclerosis (ALS): an Irish perspective. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 16(1–2), 58–62 (2015). https://doi.org/10.3109/21678421.2014.957322. (Epub 2014 Oct 6)

    Article  PubMed  Google Scholar 

  45. Gladman, M., Dharamshi, C., Zinman, L.: Economic burden of amyotrophic lateral sclerosis: a Canadian study of out-of-pocket expenses. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 15(5–6), 426–432 (2014). https://doi.org/10.3109/21678421.2014.932382. (Epub 2014 Jul 15)

    Article  PubMed  Google Scholar 

  46. Lopes de Almeida, J.P., Pinto, A., Pinto, S., Ohana, B., de Carvalho, M.: Economic cost of home-telemonitoring care for BiPAP-assisted ALS individuals. Amyotroph Lateral Scler. 13(6), 533–537 (2012). https://doi.org/10.3109/17482968.2012.703675. (Epub 2012 Aug 8)

  47. Meng, L., Bian, A., Jordan, S., Wolff, A., Shefner, J.M., Andrews, J.: Profile of medical care costs in patients with amyotrophic lateral sclerosis in the Medicare programme and under commercial insurance. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 19(1–2), 134–142 (2018). https://doi.org/10.1080/21678421.2017.1363242. (Epub 2017 Sep 11)

    Article  PubMed  Google Scholar 

  48. Pan, W., Chen, X., Bao, J., Bai, Y., Lu, H., Wang, Q., Liu, Y., Yuan, C., Li, W., Liu, Z., Liu, J., Zhu, X., Qin, B., Cai, D., Zhou, H.: The use of integrative therapies in patients with amyotrophic lateral sclerosis in shanghai, china. Evid. Based. Complement. Alternat. Med. 2013, 613596 (2013). https://doi.org/10.1155/2013/613596. (Epub 2013 Dec 2)

    Article  PubMed  PubMed Central  Google Scholar 

  49. Canadian Institute for Health, I.: The burden of neurological diseases, disorders and injuries in Canada (2007). https://secure.cihi.ca/free_products/BND_e.pdf

  50. McCabe, M.P., O’Connor, E.J.: The Economic impact of progressive neurological ilness on quality of Lige in Australia. J. Fam. Econ. Issues 31(1), 82–89 (2010)

    Article  Google Scholar 

  51. Darba, J.: Current status and direct medical cost of amyotrophic lateral sclerosis in the region of Catalonia: a population-based analysis. PLoS One 14(10), e0223772 (2019). https://doi.org/10.1371/journal.pone.0223772

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  52. Moore, A., Young, C.A., Hughes, D.A.: Health utilities and costs for motor neurone disease. Value. Health. 22(11), 1257–1265 (2019). https://doi.org/10.1016/j.jval.2019.05.011

    Article  PubMed  Google Scholar 

  53. Zwicker, J., Qureshi, D., Talarico, R., Bourque, P., Scott, M., Chin-Yee, N., Tanuseputro, P.: Dying of amyotrophic lateral sclerosis: health care use and cost in the last year of life. Neurology 93(23), e2083–e2093 (2019). https://doi.org/10.1212/WNL.0000000000008582

    Article  PubMed  Google Scholar 

  54. Wagner, L., Archer, N.P., Williamson, D.M., Henry, J.P., Schiffer, R., Jackson, C.E.: Prevalence of amyotrophic lateral sclerosis in Texas, 1998–2003. Tex. Med. 108(5), e1 (2012)

    PubMed  Google Scholar 

  55. Orphanet Report Series: prevalence of rare diseases: Bibliographic data-May 2012-Number 1 (2012).http://www.orpha.net/orphacom/cahiers/docs/GB/Prevalence_of_rare_diseases_by_alphabetical_list.pdf. Accessed June 2020

  56. ALS Society of Ontario: What is ALS? (2006) http://www.alsont.ca/what_is_als.aspx. Accessed 20 Jun 2006

  57. Alcaz, S., Jarebinski, M., Pekmezovic, T., Stevic-Marinkovic, Z., Pavlovic, S., Apostolski, S.: Epidemiological and clinical characteristics of ALS in Belgrade Yugoslavia. Acta. Neurol. Scand. 94(4), 264–268 (1996). https://doi.org/10.1111/j.1600-0404.1996.tb07063.x

    Article  CAS  PubMed  Google Scholar 

  58. Orphanet Report Series: prevalence of rare diseases: Bibliographic data-May 2011-Number 1 (2011). http://www.orpha.net/orphacom/cahiers/docs/GB/Prevalence_of_rare_diseases_by_alphabetical_list.pdf.

  59. Worms, P.M.: The epidemiology of motor neuron diseases: a review of recent studies. J. Neurol. Sci. 191(1–2), 3–9 (2001). https://doi.org/10.1016/s0022-510x(01)00630-x

    Article  CAS  PubMed  Google Scholar 

  60. Elamin, M., Phukan, J., Bede, P., Jordan, N., Byrne, S., Pender, N., Hardiman, O.: Executive dysfunction is a negative prognostic indicator in patients with ALS without dementia. Neurology 76(14), 1263–1269 (2011). https://doi.org/10.1212/WNL.0b013e318214359f

    Article  CAS  PubMed  Google Scholar 

  61. Gordon, P.H., Wang, Y., Doorish, C., Lewis, M., Battista, V., Mitsumoto, H., Marder, K.: A screening assessment of cognitive impairment in patients with ALS. Amyotroph. Lateral. Scler. 8(6), 362–365 (2007). https://doi.org/10.1080/17482960701500817

    Article  PubMed  Google Scholar 

  62. Strong, M.J., Abrahams, S., Goldstein, L.H., Woolley, S., McLaughlin, P., Snowden, J., Mioshi, E., Roberts-South, A., Benatar, M., HortobaGyi, T., Rosenfeld, J., Silani, V., Ince, P.G., Turner, M.R.: Amyotrophic lateral sclerosis-frontotemporal spectrum disorder (ALS-FTSD): revised diagnostic criteria. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 18(3–4), 153–174 (2017). https://doi.org/10.1080/21678421.2016.1267768

    Article  PubMed  PubMed Central  Google Scholar 

  63. Oecd: Health spending. https://data.oecd.org/healthres/health-spending.htm. Accessed June 2020

  64. World Health Organization: The impact of health expenditure on households and options for alternative financing (No. EM/RC51/4) (2004)

  65. World Health Organization: Global spending on health: a World in Transition. No. WHO/HIS/HGF/HFWorkingPaper/19.4 (2019)

  66. Oecd: Average wages (2020)

  67. Larg, A., Moss, J.R.: Cost-of-illness studies: a guide to critical evaluation. Pharmacoeconomics 29(8), 653–671 (2011). https://doi.org/10.2165/11588380-000000000-00000

    Article  PubMed  Google Scholar 

  68. Rooney, J.P.K., Brayne, C., Tobin, K., Logroscino, G., Glymour, M.M., Hardiman, O.: Benefits, pitfalls, and future design of population-based registers in neurodegenerative disease. Neurology 88(24), 2321–2329 (2017). https://doi.org/10.1212/WNL.0000000000004038. (Epub 2017 May 17)

    Article  PubMed  Google Scholar 

  69. Huisman, M.H., de Jong, S.W., van Doormaal, P.T., Weinreich, S.S., Schelhaas, H.J., van der Kooi, A.J., de Visser, M., Veldink, J.H., van den Berg, L.H.: Population based epidemiology of amyotrophic lateral sclerosis using capture-recapture methodology. J. Neurol. Neurosurg. Psychiatry. 82(10), 1165–1170 (2011). https://doi.org/10.1136/jnnp.2011.244939. (Epub 2011 May 27)

    Article  PubMed  Google Scholar 

  70. Traynor, B.J., Codd, M.B., Corr, B., Forde, C., Frost, E., Hardiman, O.: Incidence and prevalence of ALS in Ireland, 1995–1997: a population-based study. Neurology 52(3), 504–509 (1999). https://doi.org/10.1212/wnl.52.3.504

    Article  CAS  PubMed  Google Scholar 

  71. Hurd, M.D., Martorell, P., Delavande, A., Mullen, K.J., Langa, K.M.: Monetary costs of dementia in the United States. N. Engl. J. Med. 368(14), 1326–1334 (2013). https://doi.org/10.1056/NEJMsa1204629

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  72. Kowal, S.L., Dall, T.M., Chakrabarti, R., Storm, M.V., Jain, A.: The current and projected economic burden of Parkinson’s disease in the United States. Mov. Disord. 28(3), 311–318 (2013). https://doi.org/10.1002/mds.25292. (Epub 2013 Feb 21)

    Article  PubMed  Google Scholar 

  73. Costa, N., Ferlicoq, L., Derumeaux-Burel, H., Rapp, T., Garnault, V., Gillette-Guyonnet, S., Andrieu, S., Vellas, B., Lamure, M., Grand, A., Molinier, L.: Comparison of informal care time and costs in different age-related dementias: a review. Biomed. Res. Int. 2013, 852368 (2013). https://doi.org/10.1155/2013/852368. (Epub 2012 Dec 5)

    Article  PubMed  Google Scholar 

  74. Obermann, M., Lyon, M.: Financial cost of amyotrophic lateral sclerosis: a case study. Amyotroph. Lateral. Scler. Frontotemporal. Degener. 16(1–2), 54–57 (2015). https://doi.org/10.3109/21678421.2014.951946. (Epub 2014 Sep 23)

    Article  CAS  PubMed  Google Scholar 

  75. Moss, A.H., Casey, P., Stocking, C.B., Roos, R.P., Brooks, B.R., Siegler, M.: Home ventilation for amyotrophic lateral sclerosis patients: outcomes, costs, and patient, family, and physician attitudes. Neurology 43(2), 438–443 (1993). https://doi.org/10.1212/wnl.43.2.438

    Article  CAS  PubMed  Google Scholar 

  76. Anderson, N.H., Gluyas, C., Mathers, S., Hudson, P., Ugalde, A.: “A monster that lives in our lives”: experiences of caregivers of people with motor neuron disease and identifying avenues for support. BMJ. Support. Palliat. Care. 9(4), e27 (2019). https://doi.org/10.1136/bmjspcare-2015-001057

    Article  PubMed  Google Scholar 

  77. Oliver, D.J., Turner, M.R.: Some difficult decisions in ALS/MND. Amyotroph. Lateral. Scler. 11(4), 339–343 (2010). https://doi.org/10.3109/17482968.2010.487532

    Article  PubMed  PubMed Central  Google Scholar 

  78. Borasio, G.D., Shaw, P.J., Hardiman, O., Ludolph, A.C., Sales Luis, M.L., Silani, V., ALSSG European: Standards of palliative care for patients with amyotrophic lateral sclerosis: results of a European survey. Amyotroph. Lateral. Scler. Other. Motor. Neuron. Disord. 2(3), 159–164 (2001). https://doi.org/10.1080/146608201753275517

    Article  CAS  PubMed  Google Scholar 

  79. Oliver, D.: Ventilation in motor neuron disease: difficult decisions in difficult circumstances. Amyotroph. Lateral. Scler. Other. Motor. Neuron. Disord. 5(1), 6–8 (2004). https://doi.org/10.1080/14660820310016615

    Article  PubMed  Google Scholar 

  80. Schöffski, O. (ed.): Grundformen gesundheitsökonomischer Evaluationen, 4th edn. Springer, Berlin (2012)

    Google Scholar 

  81. Song, F., Hooper, L., Loke, Y.K.: Publication bias: what is it? How do we measure it? How do we avoid it? Open. Access. J. Clin. Trials. 2013(5), 71–81 (2013)

    Article  Google Scholar 

  82. Rosenthal, R.: The “file drawer problem” and tolerance for null results. Psychol. Bull. 86(3), 638–641 (1979)

    Article  Google Scholar 

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Acknowledgements

The authors are very grateful to Manuel Recker, who gave his valuable feedback on the systematic review.

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No funding was received from any source for the project.

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  1. Institute for Applied Health Services Research (inav GmbH), Berlin, Germany

    K. Achtert & L. Kerkemeyer

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  1. K. Achtert
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  2. L. Kerkemeyer
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KA: study rationale and design, literature search, literature selection, quality assessment of studies, interpretation and reflection, writing of the manuscript. LK: literature search, literature selection, quality assessment of studies, revising and reviewing of the manuscript.

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Correspondence to K. Achtert.

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Achtert, K., Kerkemeyer, L. The economic burden of amyotrophic lateral sclerosis: a systematic review. Eur J Health Econ 22, 1151–1166 (2021). https://doi.org/10.1007/s10198-021-01328-7

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