Elicitation of Health-Related Utility in Perianal Fistula in Crohn’s Disease

  • Louise LongworthEmail author
  • Donna Fountain
  • Jeshika Singh
  • Ismail Azzabi
  • Glynn Owen
  • Ulf Lundstam
  • Shaji Sebastian
Original Research Article


Background and Objective

Perianal fistulae are a common complication of Crohn’s disease (CD) and pose a substantial burden on quality of life. Data capturing health-related utility associated with perianal fistulae in CD are scarce. The current study aims to value health states related to different stages of the disease to quantitatively evaluate the impact of complex perianal fistulae on CD patients’ quality of life.


Eight health state descriptions associated with complex perianal fistulae in CD were developed following qualitative research with patients and validation by clinicians. Following pre-testing, a survey was administered online in two samples of UK respondents: the general population and patients with CD. A choice-based valuation technique, the time trade-off (TTO), was used for direct utility measurement. CD patients also valued their current health state using the TTO. Exclusion criteria for respondents displaying logical inconsistencies were applied.


Usable responses were received from 835 respondents, reflective of the UK population in age and sex, in the general population survey and 162 CD patients in the patient survey. Non-remission states were valued much lower than the remission state by both samples, ranging from 0.20 for proctectomy with a negative outcome to 0.66 for chronic symptomatic fistulae with mild symptoms. Patients currently experiencing fistulae reported lower values for current health than those without fistulae.


Low utility values were assigned to the non-remission health states for perianal fistulae in CD by the general public and patients with CD. This demonstrates the high humanistic burden of inadequately managed perianal fistula in CD.



The authors thank all the participants of the study. The authors also thank Persefoni Ioannou and Nicola Illingworth from PHMR for editorial assistance.

Author contributions

LL conceived the study, participated in its design and coordination, and drafted the manuscript; DF coordinated the study, participated in the design and interpretation of the data; IA participated in study design, interpretation of the data and helped to draft the study; JS performed the statistical analysis, contributed to the interpretation of the data and drafted the manuscript; GO, SS and UL contributed to the interpretation of the data and drafting of the manuscript. All authors read and approved the final manuscript.

Compliance with Ethical Standards


This research and preparation of the manuscript was funded by Takeda Pharmaceuticals.

Conflict of interest

Ismail Azzabi and Glynn Owen were employees at Takeda when this study was conducted. Shaji Sebastian has received grants and consulting fees from Takeda. Ulf Lundstarm has received consultancy fees from Takeda. Louise Longworth, Donna Fountain and Jeshika Singh declare that they have no conflict of interest.

Data availability statement

Data subject to third-party restriction. The data that support the findings of this study are available from Takeda Pharmaceuticals but restrictions apply to the availability of these data, which were used under license for the current study, and so are not publicly available. Data are, however, available from the authors upon reasonable request and with permission of Takeda Pharmaceuticals.

Supplementary material

40271_2018_352_MOESM1_ESM.docx (20 kb)
Supplementary material 1 (DOCX 20 kb)
40271_2018_352_MOESM2_ESM.docx (34 kb)
Supplementary material 2 (DOCX 33 kb)


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Copyright information

© Springer Nature Switzerland AG 2018

Authors and Affiliations

  1. 1.PHMR LimitedLondonUK
  2. 2.Takeda Pharmaceuticals International GmbHZurichSwitzerland
  3. 3.Takeda UK LtdHigh WycombeUK
  4. 4.Gothenburg UniversityGöteborgSweden
  5. 5.Hull and East Yorkshire HospitalHullUK

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