Oral human papillomavirus is common in individuals with Fanconi anemia

Sharon L. Sauter, Susanne I. Wells, Xue Zhang, Elizabeth E. Hoskins, Stella M. Davies, Kasiani C. Myers, Robin Mueller, Gitika Panicker, Elizabeth R. Unger, Umasundari Sivaprasad, Darron Brown, Parinda A. Mehta, Melinda Butsch Kovacic

Research output: Contribution to journalArticle

16 Citations (Scopus)

Abstract

Background: Fanconi anemia is a rare genetic disorder resulting in a loss of function of the Fanconi anemia-related DNA repair pathway. Individuals with Fanconi anemia are predisposed to some cancers, including oropharyngeal and gynecologic cancers, with known associations with human papillomavirus (HPV) in the general population. As individuals with Fanconi anemia respond poorly to chemotherapy and radiation, prevention of cancer is critical. Methods: To determine whether individuals with Fanconi anemia are particularly susceptible to oral HPV infection, we analyzed survey-based risk factor data and tested DNA isolated from oral rinses from 126 individuals with Fanconi anemia and 162 unaffected first-degree family members for 37 HPV types. Results: Fourteen individuals (11.1%) with Fanconi anemia tested positive, significantly more (P = 0.003) than family members (2.5%). While HPV prevalence was even higher for sexually active individuals with Fanconi anemia (17.7% vs. 2.4% in family; P = 0.003), HPV positivity also tended to be higher in the sexually inactive (8.7% in Fanconi anemia vs. 2.9% in siblings). Indeed, having Fanconi anemia increased HPV positivity 4.9-fold (95% CI, 1.6-15.4) considering age and sexual experience, but did not differ by other potential risk factors. Conclusion: Our studies suggest that oral HPV is more common in individuals with Fanconi anemia. It will be essential to continue to explore associations between risk factors and immune dysfunction on HPV incidence and persistence over time. Impact: HPV vaccination should be emphasized in those with Fanconi anemia as a first step to prevent oropharyngeal cancers, although additional studies are needed to determine whether the level of protection it offers in this population is adequate.

Original languageEnglish (US)
Pages (from-to)864-872
Number of pages9
JournalCancer Epidemiology Biomarkers and Prevention
Volume24
Issue number5
DOIs
StatePublished - May 1 2015

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Fanconi Anemia
Oropharyngeal Neoplasms
Inborn Genetic Diseases
Papillomavirus Infections
DNA Repair
Population
Siblings
Neoplasms
Vaccination

ASJC Scopus subject areas

  • Epidemiology
  • Oncology

Cite this

Sauter, S. L., Wells, S. I., Zhang, X., Hoskins, E. E., Davies, S. M., Myers, K. C., ... Kovacic, M. B. (2015). Oral human papillomavirus is common in individuals with Fanconi anemia. Cancer Epidemiology Biomarkers and Prevention, 24(5), 864-872. https://doi.org/10.1158/1055-9965.EPI-15-0097-T

Oral human papillomavirus is common in individuals with Fanconi anemia. / Sauter, Sharon L.; Wells, Susanne I.; Zhang, Xue; Hoskins, Elizabeth E.; Davies, Stella M.; Myers, Kasiani C.; Mueller, Robin; Panicker, Gitika; Unger, Elizabeth R.; Sivaprasad, Umasundari; Brown, Darron; Mehta, Parinda A.; Kovacic, Melinda Butsch.

In: Cancer Epidemiology Biomarkers and Prevention, Vol. 24, No. 5, 01.05.2015, p. 864-872.

Research output: Contribution to journalArticle

Sauter, SL, Wells, SI, Zhang, X, Hoskins, EE, Davies, SM, Myers, KC, Mueller, R, Panicker, G, Unger, ER, Sivaprasad, U, Brown, D, Mehta, PA & Kovacic, MB 2015, 'Oral human papillomavirus is common in individuals with Fanconi anemia', Cancer Epidemiology Biomarkers and Prevention, vol. 24, no. 5, pp. 864-872. https://doi.org/10.1158/1055-9965.EPI-15-0097-T
Sauter, Sharon L. ; Wells, Susanne I. ; Zhang, Xue ; Hoskins, Elizabeth E. ; Davies, Stella M. ; Myers, Kasiani C. ; Mueller, Robin ; Panicker, Gitika ; Unger, Elizabeth R. ; Sivaprasad, Umasundari ; Brown, Darron ; Mehta, Parinda A. ; Kovacic, Melinda Butsch. / Oral human papillomavirus is common in individuals with Fanconi anemia. In: Cancer Epidemiology Biomarkers and Prevention. 2015 ; Vol. 24, No. 5. pp. 864-872.
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abstract = "Background: Fanconi anemia is a rare genetic disorder resulting in a loss of function of the Fanconi anemia-related DNA repair pathway. Individuals with Fanconi anemia are predisposed to some cancers, including oropharyngeal and gynecologic cancers, with known associations with human papillomavirus (HPV) in the general population. As individuals with Fanconi anemia respond poorly to chemotherapy and radiation, prevention of cancer is critical. Methods: To determine whether individuals with Fanconi anemia are particularly susceptible to oral HPV infection, we analyzed survey-based risk factor data and tested DNA isolated from oral rinses from 126 individuals with Fanconi anemia and 162 unaffected first-degree family members for 37 HPV types. Results: Fourteen individuals (11.1{\%}) with Fanconi anemia tested positive, significantly more (P = 0.003) than family members (2.5{\%}). While HPV prevalence was even higher for sexually active individuals with Fanconi anemia (17.7{\%} vs. 2.4{\%} in family; P = 0.003), HPV positivity also tended to be higher in the sexually inactive (8.7{\%} in Fanconi anemia vs. 2.9{\%} in siblings). Indeed, having Fanconi anemia increased HPV positivity 4.9-fold (95{\%} CI, 1.6-15.4) considering age and sexual experience, but did not differ by other potential risk factors. Conclusion: Our studies suggest that oral HPV is more common in individuals with Fanconi anemia. It will be essential to continue to explore associations between risk factors and immune dysfunction on HPV incidence and persistence over time. Impact: HPV vaccination should be emphasized in those with Fanconi anemia as a first step to prevent oropharyngeal cancers, although additional studies are needed to determine whether the level of protection it offers in this population is adequate.",
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AU - Wells, Susanne I.

AU - Zhang, Xue

AU - Hoskins, Elizabeth E.

AU - Davies, Stella M.

AU - Myers, Kasiani C.

AU - Mueller, Robin

AU - Panicker, Gitika

AU - Unger, Elizabeth R.

AU - Sivaprasad, Umasundari

AU - Brown, Darron

AU - Mehta, Parinda A.

AU - Kovacic, Melinda Butsch

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N2 - Background: Fanconi anemia is a rare genetic disorder resulting in a loss of function of the Fanconi anemia-related DNA repair pathway. Individuals with Fanconi anemia are predisposed to some cancers, including oropharyngeal and gynecologic cancers, with known associations with human papillomavirus (HPV) in the general population. As individuals with Fanconi anemia respond poorly to chemotherapy and radiation, prevention of cancer is critical. Methods: To determine whether individuals with Fanconi anemia are particularly susceptible to oral HPV infection, we analyzed survey-based risk factor data and tested DNA isolated from oral rinses from 126 individuals with Fanconi anemia and 162 unaffected first-degree family members for 37 HPV types. Results: Fourteen individuals (11.1%) with Fanconi anemia tested positive, significantly more (P = 0.003) than family members (2.5%). While HPV prevalence was even higher for sexually active individuals with Fanconi anemia (17.7% vs. 2.4% in family; P = 0.003), HPV positivity also tended to be higher in the sexually inactive (8.7% in Fanconi anemia vs. 2.9% in siblings). Indeed, having Fanconi anemia increased HPV positivity 4.9-fold (95% CI, 1.6-15.4) considering age and sexual experience, but did not differ by other potential risk factors. Conclusion: Our studies suggest that oral HPV is more common in individuals with Fanconi anemia. It will be essential to continue to explore associations between risk factors and immune dysfunction on HPV incidence and persistence over time. Impact: HPV vaccination should be emphasized in those with Fanconi anemia as a first step to prevent oropharyngeal cancers, although additional studies are needed to determine whether the level of protection it offers in this population is adequate.

AB - Background: Fanconi anemia is a rare genetic disorder resulting in a loss of function of the Fanconi anemia-related DNA repair pathway. Individuals with Fanconi anemia are predisposed to some cancers, including oropharyngeal and gynecologic cancers, with known associations with human papillomavirus (HPV) in the general population. As individuals with Fanconi anemia respond poorly to chemotherapy and radiation, prevention of cancer is critical. Methods: To determine whether individuals with Fanconi anemia are particularly susceptible to oral HPV infection, we analyzed survey-based risk factor data and tested DNA isolated from oral rinses from 126 individuals with Fanconi anemia and 162 unaffected first-degree family members for 37 HPV types. Results: Fourteen individuals (11.1%) with Fanconi anemia tested positive, significantly more (P = 0.003) than family members (2.5%). While HPV prevalence was even higher for sexually active individuals with Fanconi anemia (17.7% vs. 2.4% in family; P = 0.003), HPV positivity also tended to be higher in the sexually inactive (8.7% in Fanconi anemia vs. 2.9% in siblings). Indeed, having Fanconi anemia increased HPV positivity 4.9-fold (95% CI, 1.6-15.4) considering age and sexual experience, but did not differ by other potential risk factors. Conclusion: Our studies suggest that oral HPV is more common in individuals with Fanconi anemia. It will be essential to continue to explore associations between risk factors and immune dysfunction on HPV incidence and persistence over time. Impact: HPV vaccination should be emphasized in those with Fanconi anemia as a first step to prevent oropharyngeal cancers, although additional studies are needed to determine whether the level of protection it offers in this population is adequate.

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