Advertisement

Productivity Losses and Costs in the Less-Common Systemic Autoimmune Rheumatic Diseases

  • Natalie McCormick
  • Carlo A. Marra
  • J. Antonio Aviña-Zubieta
Health Economics and Quality of Life (N Tsao, Section Editor)
Part of the following topical collections:
  1. Topical Collection on Health Economics and Quality of Life

Abstract

Purpose of Review

We synthesised the literature on productivity losses and costs in the less-common systemic autoimmune rheumatic diseases: Sjogren’s syndrome (SjS), systemic sclerosis (SSc), poly/dermatomyositis (PM/DM), and systemic vasculitides (SV).

Recent Findings

Of 29 studies located, 12 were published 2012 onwards (SSc = 6, SjS = 2, PM/DM = 2, SV = 2). In these, 25% of PM/DM, and 21–26% of SV, were work disabled, 22% of SSc stopped work within 3 years of diagnosis, and annual costs of absenteeism in SSc averaged $12,024 2017 USD. Very few studies reported on costs, presenteeism (working at reduced levels), or unpaid productivity loss. Across multiple systemic autoimmune rheumatic diseases (SARDs), major drivers of lost productivity were generalised items like pain, depression, and fatigue, rather than disease-specific factors.

Summary

Evidence suggests that work disability is common in SSc and strikes quickly. However, in SSc and other SARDs, more comprehensive estimates are needed, which include absenteeism and presenteeism from paid and unpaid work, costs, and drivers of productivity loss.

Keywords

Work disability Productivity loss Indirect costs Systemic sclerosis Sjogren’s syndrome Systemic vasculitis 

Notes

Compliance with Ethical Standards

Conflict of Interest

The authors declare that they have no conflict of interest.

Human and Animal Rights and Informed Consent

This article does not contain any studies with human or animal subjects performed by any of the authors.

References

Papers of particular interest, published recently, have been highlighted as: • Of importance •• Of major importance

  1. 1.
    Bradford Rice J, White A, Lopez A, Galebach P, Schepman P, Popelar B, et al. Healthcare resource utilization and work loss in dermatomyositis and polymyositis patients in a privately-insured US population. J Med Econ. 2016;19:649–54.CrossRefPubMedGoogle Scholar
  2. 2.
    Furst D, Amato A, Iorga S, Bancroft T, Fernandes A. Medical costs and health-care resource use in patients with inflammatory myopathies in an insured population. Muscle Nerve. 2012;46:496–505.CrossRefPubMedGoogle Scholar
  3. 3.
    Furst DE, Fernandes AW, Iorga SR, Greth W, Bancroft T. Annual medical costs and healthcare resource use in patients with systemic sclerosis in an insured population. J Rheumatol. 2012;39:2303–9.CrossRefPubMedGoogle Scholar
  4. 4.
    Furst DE, Clarke A, Fernandes AW, Bancroft T, Gajria K, Greth W, et al. Resource utilization and direct medical costs in adult systemic lupus erythematosus patients from a commercially insured population. Lupus. 2013;22:268–78.CrossRefPubMedGoogle Scholar
  5. 5.
    Raimundo K, Farr AM, Kim G, Duna G. Clinical and economic burden of antineutrophil cytoplasmic antibody-associated vasculitis in the United States. J Rheumatol. 2015;42:2383–91.CrossRefPubMedGoogle Scholar
  6. 6.
    Babigumira JB, Li M, Boudreau DM, Best JH, Garrison LP. Estimating the cost of illness of giant cell arteritis in the United States. Rheumatol Ther. 2017; Available from: http://link.springer.com/10.1007/s40744-017-0052-8Google Scholar
  7. 7.
    Yilmaz N, Can M, Oner FA, Kalfa M, Emmungil H, Karadag O, et al. Impaired quality of life, disability and mental health in Takayasu’s arteritis. Rheumatology. 2013;52:1898–904.CrossRefPubMedGoogle Scholar
  8. 8.
    Abularrage CJ, Slidell MB, Sidawy AN, Kreishman P, Amdur RL, Arora S. Quality of life of patients with Takayasu’s arteritis. J Vasc Surg. 2008;47:131–6.Google Scholar
  9. 9.
    Hudson M, Thombs BD, Steele R, Panopalis P, Newton E, Baron M, et al. Quality of life in patients with systemic sclerosis compared to the general population and patients with other chronic conditions. J Rheumatol. 2009;36:768–72.CrossRefPubMedGoogle Scholar
  10. 10.
    Jolly M. How does quality of life of patients with systemic lupus erythematosus compare with that of other common chronic illnesses? J Rheumatol. 2005;32:1706–8.PubMedGoogle Scholar
  11. 11.
    Carpenter DM, Thorpe CT, Lewis M, Devellis RF, Hogan SL. Health-related quality of life for patients with vasculitis and their spouses. Arthritis Rheum. 2009;61:259–65.CrossRefPubMedPubMedCentralGoogle Scholar
  12. 12.
    Faurschou M, Sigaard L, Bjorner JB, Baslund B. Impaired health-related quality of life in patients treated for Wegener’s granulomatosis. J Rheumatol. 2010;37:2081–5.CrossRefPubMedGoogle Scholar
  13. 13.
    Baker K, Pope J. Employment and work disability in systemic lupus erythematosus: a systematic review. Rheumatol. Oxf. Engl. 2009;48:281–4.CrossRefGoogle Scholar
  14. 14.
    Decuman S, Smith V, Verhaeghe STL, Van Hecke A, De Keyser F. Work participation in patients with systemic sclerosis: a systematic review. Clin Exp Rheumatol. 2014;32:S-206–13.Google Scholar
  15. 15.
    Poole J, Willer K, Mendelson C. Occupation of motherhood: challenges for mothers with scleroderma. Am J Occup Ther. 2009;63:214–9.CrossRefPubMedGoogle Scholar
  16. 16.
    Poole J, Rymek-Gmytrasiewicz M, Mendelson C, Sanders M, Skipper B. Parenting: the forgotten role of women living with systemic lupus erythematosus. Clin Rheumatol. 2012;31:995–1000.CrossRefPubMedGoogle Scholar
  17. 17.
    Poole JL, Willer K, Mendelson C, Sanders M, Skipper B. Perceived parenting ability and systemic sclerosis. Musculoskeletal Care. 2011;9:32–40.CrossRefPubMedGoogle Scholar
  18. 18.
    Poole JL, Chandrasekaran A, Hildebrand K, Skipper B. Participation in life situations by persons with systemic sclerosis. Disabil Rehabil. 2015;37:842–5.CrossRefPubMedGoogle Scholar
  19. 19.
    Katz P, Morris A, Trupin L, Yazdany J, Yelin E. Disability in valued life activities among individuals with systemic lupus erythematosus. Arthritis Rheum. 2008;59:465–73.CrossRefPubMedPubMedCentralGoogle Scholar
  20. 20.
    •• Mandl T, Jørgensen TS, Skougaard M, Olsson P, Kristensen L-E. Work disability in newly diagnosed patients with primary Sjögren syndrome. J Rheumatol. 2017;44:209–15. Linked data from a clinical registry, with population-based social insurance data for incident cases (SjS and SSc) and a sample of the general population, in order to assess work disability longitudinally from the time of SjS/SSc diagnosis. CrossRefPubMedGoogle Scholar
  21. 21.
    Hudson M, Steele R, Lu Y, Thombs BD, Group CSR, Baron M. Work disability in systemic sclerosis. J Rheumatol. 2009;36:2481–6.CrossRefPubMedGoogle Scholar
  22. 22.
    Bernatsky S, Hudson M, Panopalis P, Clarke AE, Pope J, Leclercq S, et al. The cost of systemic sclerosis. Arthritis Rheum. 2009;61:119–23.CrossRefPubMedGoogle Scholar
  23. 23.
    Clarke AE, Petri M, Manzi S, Isenberg DA, Gordon C, Senécal J-L, et al. The systemic lupus erythematosus tri-nation study: absence of a link between health resource use and health outcome. Rheumatology. 2004;43:1016–24.CrossRefPubMedGoogle Scholar
  24. 24.
    Clarke A, Esdaile J, Bloch B, Lacaille D, Danoff D, Fries J. A Canadian study of the total medical costs for patients with systemic lupus erythematosus and the predictors of costs. Arthritis Rheum. 1993;36:1548–59.CrossRefPubMedGoogle Scholar
  25. 25.
    Zhu TY, Tam LS, Li EK. Cost-of-illness studies in systemic lupus erythematosus: a systematic review. Arthritis Care Res. 2011;63:751–60.CrossRefGoogle Scholar
  26. 26.
    Turchetti G, Yazdany J, Palla I, Yelin E, Mosca M. Systemic lupus erythematosus and the economic perspective: a systematic literature review and points to consider. Clin Exp Rheumatol. 2012;30:S116–22.PubMedPubMedCentralGoogle Scholar
  27. 27.
    Meacock R, Dale N, Harrison MJ. The humanistic and economic burden of systemic lupus erythematosus: a systematic review. PharmacoEconomics. 2013;31:49–61.CrossRefPubMedGoogle Scholar
  28. 28.
    Panopalis P, Clarke AE, Yelin E. The economic burden of systemic lupus erythematosus. Best Pract Res Clin Rheumatol. 2012;26:695–704.CrossRefPubMedGoogle Scholar
  29. 29.
    Clarke A, Panopalis P. Systemic lupus erythematosus: clinical manifestations, treatment and economics. Expert Rev Pharmacoecon Outcomes Res. 2006;6:563–75.CrossRefPubMedGoogle Scholar
  30. 30.
    Trieste L, Palla I, Baldini C, Talarico R, D’Angiolella L, Mosca M, et al. Systemic vasculitis: how little we know about their societal and economic burden. Clin Exp Rheumatol. 2012;30:S154–6.PubMedGoogle Scholar
  31. 31.
    Schouffoer AA, Schoones JW, Terwee CB, Vliet Vlieland TPM. Work status and its determinants among patients with systemic sclerosis: a systematic review. Rheumatol Oxf Engl. 2012;51:1304–14.CrossRefGoogle Scholar
  32. 32.
    Zhang W, Bansback N, Anis AH. Measuring and valuing productivity loss due to poor health: a critical review. Soc Sci Med. 2011;72:185–92.CrossRefPubMedGoogle Scholar
  33. 33.
    Drummond MF, editor. Methods for the economic evaluation of health care programmes. 3. ed., reprint ed. Oxford: Oxford Univ. Press; 2007.Google Scholar
  34. 34.
    Larg A, Moss JR. Cost-of-illness studies: a guide to critical evaluation. PharmacoEconomics. 2011;29:653–71.CrossRefPubMedGoogle Scholar
  35. 35.
    Krol M, Brouwer W. How to estimate productivity costs in economic evaluations. PharmacoEconomics. 2014;32:335–44.CrossRefPubMedGoogle Scholar
  36. 36.
    Bowman SJ, Pierre YS, Sutcliffe N, Isenberg DA, Goldblatt F, Price E, et al. Estimating indirect costs in primary Sjogren’s syndrome. J Rheumatol. 2010;37:1010–5.CrossRefPubMedGoogle Scholar
  37. 37.
    Clarke AE, Penrod J, Pierre YS, Petri MA, Manzi S, Isenberg DA, et al. Underestimating the value of women: assessing the indirect costs of women with systemic lupus erythematosus. Tri-Nation Study Group J Rheumatol. 2000;27:2597–604.Google Scholar
  38. 38.
    Bjerrum K, Prause JU. [Sociomedical aspects of primary Sjögren’s syndrome]. Ugeskr. Laeger. 1990;152:2113–6.Google Scholar
  39. 39.
    Belotti Masserini A, Zeni S, Cossutta R, Soldi A, Fantini F. Cost-of-illness in systemic sclerosis: a retrospective study of an Italian cohort of 106 patients. Reumatismo. 2003;55:245–55.PubMedGoogle Scholar
  40. 40.
    Segal B, Bowman SJ, Fox PC, Vivino FB, Murukutla N, Brodscholl J, et al. Primary Sjogren’s syndrome: health experiences and predictors of health quality among patients in the United States. Health Qual Life Outcomes. 2009;7:46.CrossRefPubMedPubMedCentralGoogle Scholar
  41. 41.
    Meijer JM, Meiners PM, Huddleston Slater JJR, Spijkervet FKL, Kallenberg CGM, Vissink A, et al. Health-related quality of life, employment and disability in patients with Sjogren’s syndrome. Rheumatology. 2009;48:1077–82.CrossRefPubMedGoogle Scholar
  42. 42.
    Bérezné A, Seror R, Morell-Dubois S, de Menthon M, Fois E, Dzeing-Ella A, et al. Impact of systemic sclerasis on occupational and professional activity with attention to patients with digital ulcers. Arthritis Care Res. 2011;63:277–85.CrossRefGoogle Scholar
  43. 43.
    Mau W, Listing J, Huscher D, Zeidler H, Zink A. Employment across chronic inflammatory rheumatic diseases and comparison with the general population. J Rheumatol. 2005;32:721–8.PubMedGoogle Scholar
  44. 44.
    Minier T, Péntek M, Brodszky V, Ecseki A, Kárpáti K, Polgár A, et al. Cost-of-illness of patients with systemic sclerosis in a tertiary care centre. Rheumatology. 2010;49:1920–8.CrossRefPubMedGoogle Scholar
  45. 45.
    Nguyen C, Poiraudeau S, Mestre-Stanislas C, Rannou F, Bérezné A, Papelard A, et al. Employment status and socio-economic burden in systemic sclerosis: a cross-sectional survey. Rheumatol. Oxf. Engl. 2010;49:982–9.CrossRefGoogle Scholar
  46. 46.
    Ouimet JM, Pope JE, Gutmanis I, Koval J. Work disability in scleroderma is greater than in rheumatoid arthritis and is predicted by high HAQ scores. Open Rheumatol J. 2008;2:44–52.CrossRefPubMedPubMedCentralGoogle Scholar
  47. 47.
    Sandqvist G, Scheja A, Eklund M. Working ability in relation to disease severity, everyday occupations and well-being in women with limited systemic sclerosis. Rheumatol. Oxf. Engl. 2008;47:1708–11.CrossRefGoogle Scholar
  48. 48.
    Sandqvist G, Scheja A, Hesselstrand R. Pain, fatigue and hand function closely correlated to work ability and employment status in systemic sclerosis. Rheumatology. 2010;49:1739–46.CrossRefPubMedGoogle Scholar
  49. 49.
    Sharif R, Mayes MD, Nicassio PM, Gonzalez EB, Draeger H, McNearney TA, et al. Determinants of work disability in patients with systemic sclerosis: a longitudinal study of the GENISOS cohort. Semin Arthritis Rheum. 2011;41:38–47.CrossRefPubMedPubMedCentralGoogle Scholar
  50. 50.
    Wilson L. Cost-of-illness of scleroderma: the case for rare diseases. Semin Arthritis Rheum. 1997;27:73–84.CrossRefPubMedGoogle Scholar
  51. 51.
    Boomsma MM, Bijl M, Stegeman CA, Kallenberg CG, Hoffman GS, Tervaert JW. Patients’ perceptions of the effects of systemic lupus erythematosus on health, function, income, and interpersonal relationships: a comparison with Wegener’s granulomatosis. Arthritis Rheum. 2002;47:196–201.CrossRefPubMedGoogle Scholar
  52. 52.
    Boomsma MM, Stegeman CA, Tervaert JW. Comparison of Dutch and US patients’ perceptions of the effects of Wegener’s granulomatosis on health, function, income, and interpersonal relationships: comment on the article by Hoffman et al. Arthritis Rheum. 1999;42:2495–7.CrossRefPubMedGoogle Scholar
  53. 53.
    Hoffman GS, Drucker Y, Cotch MF, Locker GA, Easley K, Kwoh K. Wegener’s granulomatosis: patient-reported effects of disease on health, function, and income. Arthritis Rheum. 1998;41:2257–62.CrossRefPubMedGoogle Scholar
  54. 54.
    Reinhold-Keller E, Herlyn K, Wagner-Bastmeyer R, Gutfleisch J, Peter HH, Raspe HH, et al. Effect of Wegener’s granulomatosis on work disability, need for medical care, and quality of life in patients younger than 40 years at diagnosis. Arthritis Rheum. 2002;47:320–5.CrossRefPubMedGoogle Scholar
  55. 55.
    • Regardt M, Welin Henriksson E, Sandqvist J, Lundberg IE, Schult M-L. Work ability in patients with polymyositis and dermatomyositis: an explorative and descriptive study. Work Read Mass. 2015;53:265–77. One of the first-known publications on productivity losses in PM/DM Google Scholar
  56. 56.
    Westhoff G, Dorner T, Zink A. Fatigue and depression predict physician visits and work disability in women with primary Sjogren’s syndrome: results from a cohort study. Rheumatology. 2012;51:262–9.CrossRefPubMedGoogle Scholar
  57. 57.
    Decuman S, Smith V, Verhaeghe S, Deschepper E, Vermeiren F, Keyser FD. Work participation and work transition in patients with systemic sclerosis: a cross-sectional study. Rheumatology. 2012;51:297–304.CrossRefPubMedGoogle Scholar
  58. 58.
    Kawalec PP, Malinowski KP. The indirect costs of systemic autoimmune diseases, systemic lupus erythematosus, systemic sclerosis and sarcoidosis: a summary of 2012 real-life data from the Social Insurance Institution in Poland. Expert Rev. Pharmacoecon. Outcomes Res. 2015;15:667–73.CrossRefPubMedGoogle Scholar
  59. 59.
    • The BURQOL-RD Research Network, López-Bastida J, Linertová R, Oliva-Moreno J, Serrano-Aguilar P, Posada-de-la-Paz M, et al. Social/economic costs and health-related quality of life in patients with scleroderma in Europe. Eur J Health Econ. 2016;17:109–17. Recruited patients and their caregivers through patient organisations (instead of academic medical centres) and a rare diseases registry, and computed the productivity costs of SSc separately for seven European countries. CrossRefGoogle Scholar
  60. 60.
    Morrisroe K, Huq M, Stevens W, Rabusa C, Proudman SM, Nikpour M, et al. Determinants of unemployment amongst Australian systemic sclerosis patients: results from a multicentre cohort study. Clin Exp Rheumatol. 2016;34(Suppl 100):79–84.PubMedGoogle Scholar
  61. 61.
    •• Sandqvist G, Hesselstrand R, Petersson IF, Kristensen LE. Work disability in early systemic sclerosis: a longitudinal population-based cohort study. J Rheumatol. 2015;42:1794–800. Linked data from a clinical registry, with population-based social insurance data for incident cases (SjS and SSc) and a sample of the general population, in order to assess work disability longitudinally from the time of SjS/SSc diagnosis CrossRefPubMedGoogle Scholar
  62. 62.
    Singh MK, Clements PJ, Furst DE, Maranian P, Khanna D. Work productivity in scleroderma: analysis from the University of California, Los Angeles scleroderma quality of life study. Arthritis Care Res. 2012;64:176–83.CrossRefGoogle Scholar
  63. 63.
    Basu N, McClean A, Harper L, Amft EN, Dhaun N, Luqmani RA, et al. Markers for work disability in anti-neutrophil cytoplasmic antibody-associated vasculitis. Rheumatology. 2014;53:953–6.CrossRefPubMedGoogle Scholar
  64. 64.
    Barra LJ, Bateman EA, Rohekar S, Pagnoux C, Moradizadeh M. Assessment of work limitations and disability in systemic vasculitis. Clin Exp Rheumatol. 2016;34:S111–4.PubMedGoogle Scholar
  65. 65.
    López-Bastida J, Linertová R, Oliva-Moreno J, Posada-de-la-Paz M, Serrano-Aguilar P. Social economic costs and health-related quality of life in patients with systemic sclerosis in Spain. Arthritis Care Res. 2013Google Scholar
  66. 66.
    Chevreul K, Brigham KB, Gandré C, Mouthon L, BURQOL-RD Research Network. The economic burden and health-related quality of life associated with systemic sclerosis in France. Scand J Rheumatol. 2015;44:238–46.CrossRefPubMedGoogle Scholar
  67. 67.
    Li X, Gignac MAM, Anis AH. The indirect costs of arthritis resulting from unemployment, reduced performance, and occupational changes while at work: Med. Care. 2006;44:304–10.Google Scholar
  68. 68.
    Yelin E, Tonner C, Trupin L, Panopalis P, Yazdany J, Julian L, et al. Work loss and work entry among persons with systemic lupus erythematosus: comparisons with a national matched sample. Arthritis Rheum. 2009;61:247–58.CrossRefPubMedPubMedCentralGoogle Scholar
  69. 69.
    Nikiphorou E, Guh D, Bansback N, Zhang W, Dixey J, Williams P, et al. Work disability rates in RA. Results from an inception cohort with 24 years follow-up. Rheumatology. 2012;51:385–92.CrossRefPubMedGoogle Scholar
  70. 70.
    Hallert E, Husberg M, Kalkan A, Bernfort L. Rheumatoid arthritis is still expensive in the new decade: a comparison between two early RA cohorts, diagnosed 1996–98 and 2006–09. Scand J Rheumatol. 2016;45:371–8.CrossRefPubMedGoogle Scholar
  71. 71.
    O’Riordan R, Doran M, Connolly D. Fatigue and activity management education for individuals with systemic lupus erythematosus. Occup Ther Int. 2017;2017:1–11.CrossRefGoogle Scholar
  72. 72.
    del Pino-Sedeño T, Trujillo-Martín MM, Ruiz-Irastorza G, Cuellar-Pompa L, de Pascual-Medina AM, Serrano-Aguilar P, et al. Effectiveness of nonpharmacologic interventions for decreasing fatigue in adults with systemic lupus erythematosus: a systematic review: treating fatigue in adults with SLE. Arthritis Care Res. 2016;68:141–8.CrossRefGoogle Scholar
  73. 73.
    Strombeck BE, Theander E, Jacobsson LTH. Effects of exercise on aerobic capacity and fatigue in women with primary Sjogren’s syndrome. Rheumatology. 2007;46:868–71.CrossRefPubMedGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC 2017

Authors and Affiliations

  • Natalie McCormick
    • 1
    • 2
  • Carlo A. Marra
    • 1
    • 2
    • 3
  • J. Antonio Aviña-Zubieta
    • 2
    • 4
  1. 1.Faculty of Pharmaceutical SciencesThe University of British ColumbiaVancouverCanada
  2. 2.Arthritis Research CanadaRichmondCanada
  3. 3.School of PharmacyUniversity of OtagoDunedinNew Zealand
  4. 4.Division of Rheumatology, Department of MedicineThe University of British ColumbiaVancouverCanada

Personalised recommendations