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Estimating Direct Cost of Cystic Fibrosis Care Using Irish Registry Healthcare Resource Utilisation Data, 2008–2012

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Abstract

Background

Understanding the determinants of cost of cystic fibrosis (CF) care and health outcomes may be useful for financial planning for the delivery of CF services. Registries contain information otherwise unavailable to healthcare activity/cost monitoring systems. We estimated the direct medical cost of CF care using registry data and examined how cost was affected by patient characteristics and CF gene (CF Transmembrane Conductance Regulator [CFTR]) mutation.

Methods

Healthcare resource utilisation data (2008–2012) were obtained for CF patients enrolled with the Irish CF Registry by 2013 from linked registry and national hospitalisation database records. Mean annual hospitalisation and medication per-patient costs were estimated by demographic profile, CFTR mutation, clinical status, and CF co-morbidity, and were presented in 2014 euro values. A mixed-effects regression model was used to examine the effect of demographic, CFTR mutation, and clinical outcomes on the log10 cost of direct medical CF care.

Results

Using 4261 observations from 1100 patients, we found that the median annual total cost per patient increased over the period 2008–2012 from €12,659 to €16,852, inpatient bed-day cost increased from €14,026 to €17,332, and medication cost increased from €5863 to €12,467. Homozygous F508-CFTR mutation (class II) cost was highest and milder mutation (class IV/V) cost was 49% lower. Baseline estimated cost in 2008 for a hypothetical underweight, homozygous F508del-CFTR 6-year-old female without chronic Pseudomonas aeruginosa/Staphylococcus aureus, CF-related diabetes (CFRD) or methicillin-resistant S. aureus (MRSA), and with a poor percent predicted forced expiratory volume in 1 s (ppFEV1) was €10,113, and was €21,082 in a 25-year-old with the same hypothetical profile. Chronic P. aeruginosa infection increased baseline cost by 39%, CF co-morbidity diabetes by 18%, and frequency of pulmonary exacerbation by 15%. Underweight, declining ppFEV1, chronic S. aureus colonisation, and time also influenced cost.

Conclusions

CFTR mutation is an important factor influencing the cost of CF care. Costs differ among cohorts of CF patients eligible to access new and emerging CFTR repair therapies. These findings support the evaluation of outcome-associated cost in CFTR mutation-specific CF patient groups.

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Notes

  1. Prices of medications reimbursed through these schemes are available from https://www.sspcrs.ie/druglist/pub;JSESSIONID12=lhkZvbKllsPXHh7zzHj7mq-TjUy_mUFHUYZb_unSvOKx3wAegmuD!-295550195!1860876782.

  2. Available from the Healthcare Pricing Office.

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Acknowledgements

We thank Dr. Abhijeet Bhanegaonkar and Ellison Suthoff at Vertex Pharmaceuticals Incorporated for their editorial support during the preparation of this manuscript. The authors wish to thank the CF patients, their families and caregivers who have registered with the CFRI, the CF care teams and hospital management teams for releasing HIPE data, the National Centre for Pharmacoeconomics, the Health Service Executive’s Health Intelligence Unit and the Healthcare Pricing Office. We also wish to thank the journal’s reviewers for their thoughtful comments, which have substantially improved the quality and content of the work.

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Authors and Affiliations

  1. Cystic Fibrosis Registry of Ireland, Woodview House, UCD Belfield, Dublin 4, Ireland

    Abaigeal D. Jackson, Godfrey Fletcher, Mary Harrington & Shijun Zhou

  2. School of Natural Sciences, Zoology Building, Trinity College Dublin, Dublin 2, Ireland

    Andrew L. Jackson

  3. UCD School of Business, Room Q210, Quinn School of Business, Belfield, Dublin 4, Ireland

    Gerardine Doyle

  4. UCD School of Public Health, Physiotherapy and Sport Science, Woodview House, UCD Belfield, Dublin 4, Ireland

    Fiona Cullinane

  5. St Vincent’s University Hospital, Elm Park, Dublin 4, Ireland

    Charles Gallagher & Edward McKone

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  1. Abaigeal D. Jackson
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Contributions

A.D.J., A.L.J. and F.C. had complete access to the anonymised study dataset. A.D.J. and G.F. designed and conducted the study. A.D.J. and F.C. conducted a review of the literature and analysed the data with statistical support provided by A.L.J. G.D. provided support with the costing methodology. M.H. and S.Z. were responsible for CFRI patient recruitment and data collection. A.D.J. prepared the manuscript draft with important data interpretation and intellectual input from G.D., E.M. and C.G. All authors approved the final manuscript.

Corresponding author

Correspondence to Abaigeal D. Jackson.

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Funding

Funding support for this study was provided by Vertex Pharmaceuticals Incorporated.

Conflict of interest

A. D. Jackson, A. L. Jackson, G. Fletcher, G. Doyle, M. Harrington, S. Zhou, F. Cullinane, C. Gallagher and E. McKone declare that they have no conflicts of interest.

Data availability statement

All the relevant data is presented within the main manuscript and supplementary files. The CFRI dataset is not a freely accessible dataset. Requests for anonymous data/information from the CFRI are considered, and are subjected to a data application process. Data/information requests are considered by CFRI’s Research Committee whose decision whether to provide data or information is final.

Additional information

E. McKone: on behalf of the Cystic Fibrosis Registry of Ireland (CFRI) Executive Committee.

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Jackson, A.D., Jackson, A.L., Fletcher, G. et al. Estimating Direct Cost of Cystic Fibrosis Care Using Irish Registry Healthcare Resource Utilisation Data, 2008–2012. PharmacoEconomics 35, 1087–1101 (2017). https://doi.org/10.1007/s40273-017-0530-4

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