IDEAS home Printed from https://ideas.repec.org/a/spr/pharme/v35y2017i4d10.1007_s40273-016-0478-9.html
   My bibliography  Save this article

Economic Studies in Motor Neurone Disease: A Systematic Methodological Review

Author

Listed:
  • Alan Moore

    (Bangor University)

  • Carolyn A. Young

    (The Walton Centre NHS Trust)

  • Dyfrig A. Hughes

    (Bangor University)

Abstract

Background Motor neurone disease (MND) is a devastating condition which greatly diminishes patients’ quality of life and limits life expectancy. Health technology appraisals of future interventions in MND need robust data on costs and utilities. Existing economic evaluations have been noted to be limited and fraught with challenges. Objective The aim of this study was to identify and critique methodological aspects of all published economic evaluations, cost studies, and utility studies in MND. Methods We systematically reviewed all relevant published studies in English from 1946 until January 2016, searching the databases of Medline, EMBASE, Econlit, NHS Economic Evaluation Database (NHS EED) and the Health Economics Evaluation Database (HEED). Key data were extracted and synthesised narratively. Results A total of 1830 articles were identified, of which 15 economic evaluations, 23 cost and 3 utility studies were included. Most economic studies focused on riluzole (n = 9). Six studies modelled the progressive decline in motor function using a Markov design but did not include mutually exclusive health states. Cost estimates for a number of evaluations were based on expert opinion and were hampered by high variability and location-specific characteristics. Few cost studies reported disease-stage-specific costs (n = 3) or fully captured indirect costs. Utilities in three studies of MND patients used the EuroQol EQ-5D questionnaire or standard gamble, but included potentially unrepresentative cohorts and did not consider any health impacts on caregivers. Conclusion Economic evaluations in MND suffer from significant methodological issues such as a lack of data, uncertainty with the disease course and use of inappropriate modelling framework. Limitations may be addressed through the collection of detailed and representative data from large cohorts of patients.

Suggested Citation

  • Alan Moore & Carolyn A. Young & Dyfrig A. Hughes, 2017. "Economic Studies in Motor Neurone Disease: A Systematic Methodological Review," PharmacoEconomics, Springer, vol. 35(4), pages 397-413, April.
    • Handle: RePEc:spr:pharme:v:35:y:2017:i:4:d:10.1007_s40273-016-0478-9
    DOI: 10.1007/s40273-016-0478-9
    as

    Download full text from publisher

    File URL: http://link.springer.com/10.1007/s40273-016-0478-9
    File Function: Abstract
    Download Restriction: Access to the full text of the articles in this series is restricted.
    File URL: https://libkey.io/10.1007/s40273-016-0478-9?utm_source=ideas
    LibKey link: if access is restricted and if your library uses this service, LibKey will redirect you to where you can use your library subscription to access this item
    ---><---

    As the access to this document is restricted, you may want to search for a different version of it.

    References listed on IDEAS

    as
    1. Annie Hawton & James Shearer & Elizabeth Goodwin & Colin Green, 2013. "Squinting Through Layers of Fog: Assessing the Cost Effectiveness of Treatments for Multiple Sclerosis," Applied Health Economics and Health Policy, Springer, vol. 11(4), pages 331-341, August.
    Full references (including those not matched with items on IDEAS)

    Most related items

    These are the items that most often cite the same works as this one and are cited by the same works as this one.
    1. A. J. Hawton & C. Green, 2016. "Multiple sclerosis: relapses, resource use, and costs," The European Journal of Health Economics, Springer;Deutsche Gesellschaft für Gesundheitsökonomie (DGGÖ), vol. 17(7), pages 875-884, September.
    2. Shien Guo & Christopher Pelligra & Catherine Saint-Laurent Thibault & Luis Hernandez & Anuraag Kansal, 2014. "Cost-Effectiveness Analyses in Multiple Sclerosis: A Review of Modelling Approaches," PharmacoEconomics, Springer, vol. 32(6), pages 559-572, June.

    Corrections

    All material on this site has been provided by the respective publishers and authors. You can help correct errors and omissions. When requesting a correction, please mention this item's handle: RePEc:spr:pharme:v:35:y:2017:i:4:d:10.1007_s40273-016-0478-9. See general information about how to correct material in RePEc.

    If you have authored this item and are not yet registered with RePEc, we encourage you to do it here. This allows to link your profile to this item. It also allows you to accept potential citations to this item that we are uncertain about.

    If CitEc recognized a bibliographic reference but did not link an item in RePEc to it, you can help with this form .

    If you know of missing items citing this one, you can help us creating those links by adding the relevant references in the same way as above, for each refering item. If you are a registered author of this item, you may also want to check the "citations" tab in your RePEc Author Service profile, as there may be some citations waiting for confirmation.

    For technical questions regarding this item, or to correct its authors, title, abstract, bibliographic or download information, contact: Sonal Shukla or Springer Nature Abstracting and Indexing (email available below). General contact details of provider: http://www.springer.com .

    Please note that corrections may take a couple of weeks to filter through the various RePEc services.

    IDEAS is a RePEc service. RePEc uses bibliographic data supplied by the respective publishers.