, Volume 33, Issue 2, pp 123–135 | Cite as

Does Including Informal Care in Economic Evaluations Matter? A Systematic Review of Inclusion and Impact of Informal Care in Cost-Effectiveness Studies

  • Marieke KrolEmail author
  • Jocé Papenburg
  • Job van Exel
Systematic Review



Informal care makes an important contribution to societal welfare. However, it may involve substantial time costs and can have a considerable negative effect on the health and well-being of informal caregivers. These costs and effects of informal caregiving are often excluded in economic evaluations of healthcare interventions. The impact of this exclusion on the outcomes of these evaluations is largely unknown.


This study aimed to explore the inclusion of informal care in economic evaluations and the potential impact of the costs and effects of informal caregiving on cost-effectiveness outcomes.


A systematic review was conducted to identify economic evaluations of interventions in four distinct disease areas where informal care is potentially important: Alzheimer’s disease, metastatic colorectal cancer, Parkinson’s disease and rheumatoid arthritis. It was recorded how often economic evaluations included informal caregiving. Next, for the studies including informal care, the impact on cost-effectiveness outcomes was determined by removing informal care costs and effects of the cost-effectiveness calculations and recalculating the outcomes. The new cost-effectiveness outcomes were then compared with the original reported outcomes.


The study identified 100 economic evaluations investigating interventions targeted at Alzheimer’s disease (n = 25), metastatic colorectal cancer (n = 24), Parkinson’s disease (n = 8) and rheumatoid arthritis (n = 43). Twenty-three of these evaluations (23 %) included costs and/or effects of informal caregiving: 64 % of the Alzheimer’s disease studies, 0 % of the metastatic colorectal cancer studies, 13 % of Parkinson’s disease studies and 14 % of rheumatoid arthritis studies. When informal care was included, this mostly concerned time costs. Studies rarely included both costs and effects. The effect of including or excluding informal care costs or effects on cost-effectiveness outcomes in most studies was modest, but in some studies the impact was strong.


Most economic evaluations in the area of Alzheimer’s disease include costs and/or effects related to informal caregiving. However, in other disease areas where informal caregiving is common it seems that the majority of economic evaluations ignore informal caregiving. The inclusion of informal care can have a strong impact on cost-effectiveness outcomes. Future economic evaluations should therefore consider the relevance of informal care in the context of their study, and either include these costs and effects or justify why they were excluded.


Economic Evaluation Time Cost Metastatic Colorectal Cancer Informal Care Informal Caregiving 
These keywords were added by machine and not by the authors. This process is experimental and the keywords may be updated as the learning algorithm improves.



This study is part of a larger project investigating the broader societal benefits of healthcare, which was financially supported by AstraZeneca, GlaxoSmithKline, Janssen, MSD BV, Novartis and Pfizer. The views expressed in this paper are those of the authors. The authors have no conflicts of interest to declare.

Author contributions

MK was primarily responsible for writing the manuscript in close cooperation with JP and JvE. The search strategies conducted in the review were the result of joint effort. MK and JP conducted the actual review process. All authors read, edited and approved the final manuscript. MK is the overall guarantor for the content.


  1. 1.
    Drummond MF, Sculpher MJ, Torrance GW, O’Brien BJ, Stoddart GL. Methods for the economic evaluation of health care programmes. 3rd ed. Oxford/New York: Oxford University Press; 2005.Google Scholar
  2. 2.
    Basu A, Meltzer D. Implications of spillover effects within the family for medical cost-effectiveness analysis. J Health Econ. 2005;24:751–73.PubMedCrossRefGoogle Scholar
  3. 3.
    Christakis NA. Social networks and collateral health effects. BMJ. 2004;329:184–5.PubMedCentralPubMedCrossRefGoogle Scholar
  4. 4.
    Bobinac A, van Exel NJ, Rutten FF, Brouwer WB. Caring for and caring about: disentangling the caregiver effect and the family effect. J Health Econ. 2010;29:549–56.PubMedCrossRefGoogle Scholar
  5. 5.
    Bobinac A, van Exel NJ, Rutten FF, Brouwer WB. Health effects in significant others: separating family and care-giving effects. Med Decis Making. 2011;31:292–8.PubMedCrossRefGoogle Scholar
  6. 6.
    Hoefman RJ, van Exel J, Brouwer W. How to include informal care in economic evaluations. Pharmacoeconomics. 2013;31:1105–19.PubMedCrossRefGoogle Scholar
  7. 7.
    Schulz R, Beach SR. Caregiving as a risk factor for mortality: the Caregiver Health Effects Study. JAMA. 1999;282:2215–9.PubMedCrossRefGoogle Scholar
  8. 8.
    Pitsenberger DJ. Juggling work and elder caregiving: work-life balance for aging American workers. AAOHN J. 2006;54:181–5 (quiz 186–7).PubMedGoogle Scholar
  9. 9.
    van Ryn M, Sanders S, Kahn K, van Houtven C, Griffin JM, Martin M, et al. Objective burden, resources, and other stressors among informal cancer caregivers: a hidden quality issue? Psychooncology. 2011;20:44–52.PubMedCrossRefGoogle Scholar
  10. 10.
    Brouwer WB. Too important to ignore: informal caregivers and other significant others. Pharmacoeconomics. 2006;24:39–41.PubMedCrossRefGoogle Scholar
  11. 11.
    Dixon S, Walker M, Salek S. Incorporating carer effects into economic evaluation. Pharmacoeconomics. 2006;24:43–53.PubMedCrossRefGoogle Scholar
  12. 12.
    Davidson T, Levin LA. Is the societal approach wide enough to include relatives? Incorporating relatives’ costs and effects in a cost-effectiveness analysis. Appl Health Econ Health Policy. 2010;8:25–35.PubMedCrossRefGoogle Scholar
  13. 13.
    Al-Janabi H, Flynn TN, Coast J. QALYs and carers. Pharmacoeconomics. 2011;29:1015–23.PubMedCrossRefGoogle Scholar
  14. 14.
    Koopmanschap MA, van Exel JN, van den Berg B, Brouwer WB. An overview of methods and applications to value informal care in economic evaluations of healthcare. Pharmacoeconomics. 2008;26:269–80.PubMedCrossRefGoogle Scholar
  15. 15.
    Goodrich K, Kaambwa B, Al-Janabi H. The inclusion of informal care in applied economic evaluation: a review. Value Health. 2012;15:975–81.PubMedCrossRefGoogle Scholar
  16. 16.
    Opara JA. Activities of daily living and quality of life in Alzheimer disease. J Med Life. 2012;5:162–7.PubMedCentralPubMedGoogle Scholar
  17. 17.
    Schrag A, Hovris A, Morley D, Quinn N, Jahanshahi M. Caregiver-burden in Parkinson’s disease is closely associated with psychiatric symptoms, falls, and disability. Parkinsonism Relat Disord. 2006;12:35–41.PubMedCrossRefGoogle Scholar
  18. 18.
    Sansoni J, Anderson KH, Varona LM, Varela G. Caregivers of Alzheimer’s patients and factors influencing institutionalization of loved ones: some considerations on existing literature. Ann Ig. 2013;25:235–46.PubMedGoogle Scholar
  19. 19.
    Birnbaum H, Pike C, Kaufman R, Marynchenko M, Kidolezi Y, Cifaldi M. Societal cost of rheumatoid arthritis patients in the US. Curr Med Res Opin. 2010;26:77–90.PubMedCrossRefGoogle Scholar
  20. 20.
    Brouwer WB, van Exel NJ, van de Berg B, Dinant HJ, Koopmanschap MA, van den Bos GA. Burden of caregiving: evidence of objective burden, subjective burden, and quality of life impacts on informal caregivers of patients with rheumatoid arthritis. Arthritis Rheum. 2004;51:570–7.PubMedCrossRefGoogle Scholar
  21. 21.
    Dalal KM, Gollub MJ, Miner TJ, Wong WD, Gerdes H, Schattner MA, et al. Management of patients with malignant bowel obstruction and stage IV colorectal cancer. J Palliat Med. 2011;14:822–8.PubMedCrossRefGoogle Scholar
  22. 22.
    Gao W, Gulliford M, Bennett MI, Murtagh FE, Higginson IJ. Managing cancer pain at the end of life with multiple strong opioids: a population-based retrospective cohort study in primary care. PLoS One. 2014;9:e79266.PubMedCentralPubMedCrossRefGoogle Scholar
  23. 23.
    Stone PW, Chapman RH, Sandberg EA, Liljas B, Neumann PJ. Measuring costs in cost-utility analyses. Variations in the literature. Int J Technol Assess Health Care. 2000;16:111–24.PubMedCrossRefGoogle Scholar
  24. 24.
    Hoefman RJ, Van Exel NJA, Brouwer WBF. iMTA Valuation of Informal Care Questionnaire (iVICQ). Version 1.0 (December 2011). Rotterdam: iBMG/iMTA; 2011. Accessed 12 Apr 2014.
  25. 25.
    Rive B, Aarsland D, Grishchenko M, Cochran J, Lamure M, Toumi M. Cost-effectiveness of memantine in moderate and severe Alzheimer’s disease in Norway. Int J Geriatr Psychiatry. 2012;27:573–82.PubMedCrossRefGoogle Scholar
  26. 26.
    Meeuwsen E, Melis R, van der Aa G, Goluke-Willemse G, de Leest B, van Raak F, et al. Cost-effectiveness of one year dementia follow-up care by memory clinics or general practitioners: economic evaluation of a randomised controlled trial. PLoS One. 2013;8:e79797.PubMedCentralPubMedCrossRefGoogle Scholar
  27. 27.
    Touchon J, Lachaine J, Beauchemin C, Granghaud A, Rive B, Bineau S. The impact of memantine in combination with acetylcholinesterase inhibitors on admission of patients with Alzheimer’s disease to nursing homes: cost-effectiveness analysis in France. Eur J Health Econ. Epub 2013 Aug 9.Google Scholar
  28. 28.
    Getsios D, Blume S, Ishak KJ, Maclaine G, Hernandez L. An economic evaluation of early assessment for Alzheimer’s disease in the United Kingdom. Alzheimers Dement. 2012;8:22–30.PubMedCrossRefGoogle Scholar
  29. 29.
    Pfeil AM, Kressig RW, Szucs TD. Alzheimer’s dementia: budget impact and cost-utility analysis of a combination treatment of a cholinesterase inhibitor and memantine in Switzerland. Swiss Med Wkly. 2012;142:w13676.PubMedGoogle Scholar
  30. 30.
    López-Bastida J, Hart W, Garcia-Perez L, Linertova R. Cost-effectiveness of donepezil in the treatment of mild or moderate Alzheimer’s disease. J Alzheimers Dis. 2009;16:399–407.PubMedGoogle Scholar
  31. 31.
    Zhang Y, Kivipelto M, Solomon A, Wimo A. Cost-effectiveness of a health intervention program with risk reductions for getting demented: results of a Markov model in a Swedish/Finnish setting. J Alzheimers Dis. 2011;26:735–44.PubMedGoogle Scholar
  32. 32.
    Hoogveldt B, Rive B, Severens J, Maman K, Guilhaume C. Cost-effectiveness analysis of memantine for moderate-to-severe Alzheimer’s disease in The Netherlands. Neuropsychiatr Dis Treat. 2011;7:313–7.PubMedCentralPubMedGoogle Scholar
  33. 33.
    Biasutti M, Dufour N, Ferroud C, Dab W, Temime L. Cost-effectiveness of magnetic resonance imaging with a new contrast agent for the early diagnosis of Alzheimer’s disease. PLoS One. 2012;7:e35559.PubMedCentralPubMedCrossRefGoogle Scholar
  34. 34.
    van den Hout WB, Goekoop-Ruiterman YP, Allaart CF, de Vries-Bouwstra JK, Hazes JM, Kerstens PJ, et al. Cost-utility analysis of treatment strategies in patients with recent-onset rheumatoid arthritis. Arthritis Rheum. 2009;61:291–9.PubMedCrossRefGoogle Scholar
  35. 35.
    Lekander I, Borgstrom F, Svarvar P, Ljung T, Carli C, van Vollenhoven RF. Cost-effectiveness of real-world infliximab use in patients with rheumatoid arthritis in Sweden. Int J Technol Assess Health Care. 2010;26:54–61.PubMedCrossRefGoogle Scholar
  36. 36.
    Ungar WJ, Costa V, Hancock-Howard R, Feldman BM, Laxer RM. Cost-effectiveness of biologics in polyarticular-course juvenile idiopathic arthritis patients unresponsive to disease-modifying antirheumatic drugs. Arthritis Care Res. 2011;63:111–9.CrossRefGoogle Scholar
  37. 37.
    Kobelt G, Lekander I, Lang A, Raffeiner B, Botsios C, Geborek P. Cost-effectiveness of etanercept treatment in early active rheumatoid arthritis followed by dose adjustment. Int J Technol Assess Health Care. 2011;27:193–200.PubMedCrossRefGoogle Scholar
  38. 38.
    Lekander I, Borgstrom F, Lysholm J, van Vollenhoven RF, Lindblad S, Geborek P, et al. The cost-effectiveness of TNF-inhibitors for the treatment of rheumatoid arthritis in Swedish clinical practice. Eur J Health Econ. 2013;14:863–73.PubMedCrossRefGoogle Scholar
  39. 39.
    Lindgren P, Geborek P, Kobelt G. Modeling the cost-effectiveness of treatment of rheumatoid arthritis with rituximab using registry data from Southern Sweden. Int J Technol Assess Health Care. 2009;25:181–9.PubMedCrossRefGoogle Scholar
  40. 40.
    Hartz S, Getsios D, Tao S, Blume S, Maclaine G. Evaluating the cost effectiveness of donepezil in the treatment of Alzheimer’s disease in Germany using discrete event simulation. BMC Neurol. 2012;12:2.PubMedCentralPubMedCrossRefGoogle Scholar
  41. 41.
    Bond M, Rogers G, Peters J, Anderson R, Hoyle M, Miners A, et al. The effectiveness and cost-effectiveness of donepezil, galantamine, rivastigmine and memantine for the treatment of Alzheimer’s disease (review of Technology Appraisal no. 111): a systematic review and economic model. Health Technol Assess. 2012;16:1–470.PubMedCrossRefGoogle Scholar
  42. 42.
    Woods RT, Bruce E, Edwards RT, Elvish R, Hoare Z, Hounsome B, et al. REMCARE: reminiscence groups for people with dementia and their family caregivers—effectiveness and cost-effectiveness pragmatic multicentre randomised trial. Health Technol. Assess. 2012;16:v–xv (1–116).PubMedCrossRefGoogle Scholar
  43. 43.
    Banerjee S, Hellier J, Romeo R, Dewey M, Knapp M, Ballard C, et al. Study of the use of antidepressants for depression in dementia: the HTA-SADD trial—a multicentre, randomised, double-blind, placebo-controlled trial of the clinical effectiveness and cost-effectiveness of sertraline and mirtazapine. Health Technol Assess. 2013;17:1–166.PubMedCrossRefGoogle Scholar
  44. 44.
    Nagy B, Brennan A, Brandtmuller A, Thomas SK, Sullivan SD, Akehurst R. Assessing the cost-effectiveness of the rivastigmine transdermal patch for Alzheimer’s disease in the UK using MMSE- and ADL-based models. Int J Geriatr Psychiatry. 2011;26:483–94.PubMedCrossRefGoogle Scholar
  45. 45.
    Romeo R, Knapp M, Hellier J, Dewey M, Ballard C, Baldwin R, et al. Cost-effectiveness analyses for mirtazapine and sertraline in dementia: randomised controlled trial. Br J Psychiatry. 2013;202:121–8.PubMedCrossRefGoogle Scholar
  46. 46.
    Haycox A, Armand C, Murteira S, Cochran J, Francois C. Cost effectiveness of rasagiline and pramipexole as treatment strategies in early Parkinson’s disease in the UK setting: an economic Markov model evaluation. Drugs Aging. 2009;26:791–801.PubMedCrossRefGoogle Scholar
  47. 47.
    Suh GH. Modeling the cost-effectiveness of galantamine for mild to moderately severe Alzheimer’s disease in Korea. Value Health. 2009;12:S49–54.PubMedCrossRefGoogle Scholar

Copyright information

© Springer International Publishing Switzerland 2014

Authors and Affiliations

  1. 1.Department of Health Policy and ManagementErasmus UniversityRotterdamThe Netherlands
  2. 2.Institute for Medical Technology AssessmentErasmus UniversityRotterdamThe Netherlands
  3. 3.Independent ResearcherSt. WillibrordusCuracao

Personalised recommendations