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J Crohns Colitis. 2016 April; 10(4): 504–505.
Published online 2015 December 30. doi:  10.1093/ecco-jcc/jjv236
PMCID: PMC4946760

Oral Cancer in a Patient Receiving Mercaptopurine for Ulcerative Colitis

Dear Editor

We would like to comment on the article titled ‘Oral Cancer and Precancerous Lesions in Inflammatory Bowel Diseases: A Systematic Review’ by Katsanos et al.1 The article summarizes literature on oral cancers in patients with inflammatory bowel disease taking azathioprine, infliximab and adalimumab. Oral cancer progression has also been reported with therapeutic doses of methotrexate and etanercept used for management of rheumatoid arthritis.2 We would like to describe a case of oral cancer associated with mercaptopurine therapy for ulcerative colitis.

A tissue sample taken from the right lateral tongue of a 24-year-old female was submitted for histopathological analysis to the oral pathology service of the Virginia Commonwealth University School of Dentistry in Richmond, Virginia. The accompanying history suggested that the patient was seen by an oral surgeon for a 3-month history of right tongue discomfort. Her past medical history was significant for ulcerative colitis diagnosed approximately 4 years previously and was being managed for 2 years by a daily dose of 75mg of mercaptopurine. The patient denied use of tobacco products, excessive alcohol and substance abuse. Clinical examination revealed no significant extra-oral findings. Intra-orally, there was a 3.5cm × 4cm ulcerative lesion on the right infero-ventral tongue (Figure 1) with indurated borders and tenderness to touch. The clinician biopsied the lesion at that visit, which was read as squamous cell carcinoma not associated with human papillomavirus (HPV) by the oral pathology service. Results were conveyed to the referring clinician, and the patient was subsequently referred to a head and neck cancer surgeon.

Figure 1.
4cm × 3.5cm ulcerative lesion on the right inferoventral tongue with indurated, elevated and irregular borders, surrounded by erythematous and hyperkeratotic areas on the tongue.

This case presentation is unique in that the patient did not possess the traditional risk factors associated with squamous cell carcinomas, such as male sex, alcoholism and tobacco use.3 Head and neck cancers linked to HPV may be encountered in younger patients, such as the one reported. However, they are usually associated with cancers of the oropharynx and the base of the tongue, and their numbers have demonstrably risen in recent decades.4 Oral lichen planus is associated with less than 2% risk of cancer transformation, as mentioned in the original article.1 Immunosuppressed states such as graft-versus-host disease5 and HIV1 are also associated with the risk of oral carcinogenesis.

As concluded by the authors of the original article, we agree that inflammatory bowel disease sufferers taking immunosuppressants should be considered at an increased risk of oral carcinogenesis, and periodic evaluation by dentists, oral surgeons, oral medicine specialists or oral pathologists is to be recommended. We would like to thank Kostanos and colleagues for their excellent review and the Journal of Crohn’s and Colitis for publishing it.

Sincerely,

Funding

No external funding sources are relevant to the contents of this submission.

Conflict of Interest

Neither author has any disclosures or conflicts of interest to report.

Author Contributions

J. Svirsky was the recipient of the biopsy and clinical history of this case and reviewed the letter subsequently. B. Desai wrote the letter, communicated with the referring provider, performed the literature search and submitted the letter to the journal.

References

1. Katsanos KH, Roda G, Brygo A, Delaporte E, Colombel JF. Oral cancer and oral precancerous lesions in inflammatory bowel diseases: a systematic review. J Crohns Colitis 2015;9:1043–52. [PubMed]
2. Chainani-wu N, Chang C, Gross AJ, Yom SS, Silverman S. Oropharyngeal carcinoma arising after methotrexate and etanercept therapy for rheumatoid arthritis. Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e261–3. [PubMed]
3. Gillison ML, D’Souza G, Westra W, et al. Distinct risk factor profiles for human papillomavirus type 16-positive and human papillomavirus type 16-negative head and neck cancers. J Natl Cancer Inst 2008;100:407–20. [PubMed]
4. Young D, Xiao CC, Murphy B, Moore M, Fakhry C, Day TA. Increase in head and neck cancer in younger patients due to human papillomavirus (HPV). Oral Oncol 2015;51:727–30. [PubMed]
5. Petti S, Polimeni A, Berloco PB, Scully C. Orofacial diseases in solid organ and hematopoietic stem cell transplant recipients. Oral Dis 2013;19:18–36. [PubMed]

Articles from Journal of Crohn's & Colitis are provided here courtesy of Oxford University Press