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Systematic review and evidence synthesis of non-cervical human papillomavirus-related disease health system costs and quality of life estimates
  1. Koh Jun Ong1,
  2. Marta Checchi2,
  3. Lorna Burns3,
  4. Charlotte Pavitt4,
  5. Maarten J Postma5,6,7,
  6. Mark Jit2,8
  1. 1 National Infection Service, Public Health England, London, UK
  2. 2 National Infection Service, Public Health England, London, UK
  3. 3 Faculty of Medicine and Dentistry, University of Plymouth, Devon, UK
  4. 4 National Infection Service, Public Health England, London, UK
  5. 5 Department of Pharmacy, University of Groningen, Groningen, The Netherlands
  6. 6 Department of Health Sciences, University Medical Center Groningen, Groningen, The Netherlands
  7. 7 Department of Economics, Econometrics & Finance, University of Groningen, Groningen, The Netherlands
  8. 8 Department of Infectious Disease Epidemiology, London School of Hygiene & Tropical Medicine, London, UK
  1. Correspondence to Mark Jit; Mark.Jit{at}


Background Many economic evaluations of human papillomavirus vaccination should ideally consider multiple disease outcomes, including anogenital warts, respiratory papillomatosis and non-cervical cancers (eg, anal, oropharyngeal, penile, vulvar and vaginal cancers). However, published economic evaluations largely relied on estimates from single studies or informal rapid literature reviews.

Methods We conducted a systematic review of articles up to June 2016 to identify costs and utility estimates admissible for an economic evaluation from a single-payer healthcare provider’s perspective. Meta-analyses were performed for studies that used same utility elicitation tools for similar diseases. Costs were adjusted to 2016/2017 US$.

Results Sixty-one papers (35 costs; 24 utilities; 2 costs and utilities) were selected from 10 742 initial records. Cost per case ranges were US$124–US$883 (anogenital warts), US$6912–US$52 579 (head and neck cancers), US$12 936–US$51 571 (anal cancer), US$17 524–34 258 (vaginal cancer), US$14 686–US$28 502 (vulvar cancer) and US$9975–US$27 629 (penile cancer). The total cost for 14 adult patients with recurrent respiratory papillomatosis was US$137 601 (one paper).

Utility per warts episode ranged from 0.651 to 1 (12 papers, various utility elicitation methods), with pooled mean EQ-5D and EQ-VAS of 0.86 (95% CI 0.85 to 0.87) and 0.74 (95% CI 0.74 to 0.75), respectively. Fifteen papers reported utilities in head and neck cancers with range 0.29 (95% CI 0.0 to 0.76) to 0.94 (95% CI 0.3 to 1.0). Mean utility reported ranged from 0.5 (95% CI 0.4 to 0.61) to 0.65 (95% CI 0.45 to 0.75) (anal cancer), 0.59 (95% CI 0.54 to 0.64) (vaginal cancer), 0.65 (95% CI 0.60 to 0.70) (vulvar cancer) and 0.79 (95% CI 0.74 to 0.84) (penile cancer).

Conclusions Differences in values reported from each paper reflect variations in cancer site, disease stages, study population, treatment modality/setting and utility elicitation methods used. As patient management changes over time, corresponding effects on both costs and utility need to be considered to ensure health economic assumptions are up-to-date and closely reflect the case mix of patients.

  • HPV
  • vaccination
  • cost-effectiveness
  • systematic reviews
  • meta-analysis

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  • Handling editor Alec Miners

  • Contributors KJO, MJP and MJ conceived and planned the systematic review. LB conducted the systematic literature searches. KJO, MC and CP carried out sifting and data extraction of the systematic literature search results. KJO conducted the meta-analysis and took the lead in writing the manuscript, with guidance from MJP and MJ. All authors provided critical feedback on the manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests MJ reports grants from the National Institute for Health Research during the conduct of the study.

  • Patient consent Not required.

  • Provenance and peer review Not commissioned; externally peer reviewed.