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PharmacoEconomics

, Volume 37, Issue 1, pp 45–61 | Cite as

Systematic Literature Review of the Economic Burden of Celiac Disease

  • Elizabeth S. MearnsEmail author
  • Aliki Taylor
  • Talia Boulanger
  • Kelly J. Craig
  • Michele Gerber
  • Daniel A. Leffler
  • Jennifer Drahos
  • David S. Sanders
  • Benjamin Lebwohl
Systematic Review

Abstract

Background

The prevalence of celiac disease (CD) has rapidly increased over recent decades, but costs related to CD remain poorly quantified.

Objective

This systematic review assessed the economic burden of CD in North America and Europe.

Methods

MEDLINE, EMBASE, EconLit, and the Cochrane Library databases were systematically searched to identify English-language literature from 2007 to 2018 that assessed costs, cost effectiveness, and health resource utilization for CD.

Results

Forty-nine studies met the inclusion criteria, of which 28 (57.1%) addressed costs of testing and diagnosis; 33 (67.3%) were from Europe. The cost per positive CD diagnosis of testing patients already undergoing esophagogastroduodenoscopy for other indications ranged from 1300 Canadian dollars ($Can) in Canada (2016 value) to €44,712 in the Netherlands (2013 value). Adding the CD test was cost effective when it combined diagnostic modalities (e.g., serology and biopsy). Direct annual excess costs to a US payer per diagnosed CD patient totaled $US6000 (2013 value) more than for a person without CD, chiefly due to outpatient care. Hospitalizations, emergency visits, and medication use were more common with CD. After initiating a gluten-free diet (GFD), patients visited primary care providers less often, used more medications, and missed fewer days from school and work.

Conclusions

Most of the few available economic studies of CD assess testing and diagnosis costs, especially in Europe. Methods of testing generally are considered cost effective when they combine diagnostic modalities in symptomatic patients. Most costs to a payer of managing CD derive from outpatient care. Following GFD initiation, patients lose fewer days from work and school than pretreatment.

Notes

Acknowledgements

We would like to thank Dr Nicole Fusco for careful review of the manuscript.

Author contributions

Guarantor of the article: Dr. Elizabeth Mearns. Authorship contributions: ESM, AT, TB, MG, and DAL contributed to the concept and design of the research study. ESM, TB, and KJC performed the research and collected the data. ESM, TB, and KJC analyzed the data. ESM, TB, and KJC wrote the paper. All authors provided critical analyses of the manuscript drafts and approved the final version of the article.

Compliance with Ethical Standards

Funding

ESM, TB, and KJC were/are employees of Truven Health Analytics, an IBM Company, and received a research contract from Takeda Pharmaceuticals International Co. to conduct this study.

Conflict of interest

ESM, TB, and KJC were/are employees of Truven Health Analytics, an IBM Company, and received a research contract from Takeda Pharmaceuticals International Co. to conduct this study. AT, MG, JD, and DAL are employed by Takeda Pharmaceuticals International Co. BL and DSS have served as consultants for Takeda Pharmaceuticals International Co.

Data availability statement

Data-sharing information is not applicable to this study as no datasets were generated or analyzed during the study.

Ethical standards

All authors complied with the Committee on Publication Ethics (COPE) guidelines to uphold the integrity of the scientific record.

Ethical approval

Not applicable.

Supplementary material

40273_2018_707_MOESM1_ESM.docx (16 kb)
Supplementary material 1 (DOCX 15 kb)

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Copyright information

© Springer Nature Switzerland AG 2018

Authors and Affiliations

  1. 1.ZS AssociatesNew YorkUSA
  2. 2.Takeda Development Centre EuropeLondonUK
  3. 3.BostonUSA
  4. 4.Truven Health Analytics, An IBM CompanyCambridgeUSA
  5. 5.Takeda Pharmaceuticals International CoCambridgeUSA
  6. 6.Royal Hallamshire Hospital and University of SheffieldSheffieldUK
  7. 7.Department of MedicineCeliac Disease Center, Columbia University Medical CenterNew YorkUSA

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