Gingival Carcinoma in a Patient With Ulcerative Colitis?

Gingival Carcinoma in a Patient With Ulcerative Colitis?

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Gingival Carcinoma in a Patient With Ulcerative Colitis? Q2

Takeshi Shiraishi,1 Hiroyuki Shoji,2 and Yuko Akazawa2 1

Department of Regenerative Oral Surgery, Unit of Translational Medicine, Nagasaki University Graduate School of Biomedical Sciences, and 2Department of Gastroenterology and Hepatology, Nagasaki University Hospital, Nagasaki, Japan

Question: A 69-year-old Japanese man was referred to the Department of Oral and Maxillofacial Surgery in our hospital because of a chronic oral ulcer in the gingiva, which was initially suspected to be a gingival carcinoma. He had been administered mesalamine and corticosteroids for the treatment of ulcerative colitis over the past 5 years. He experienced spontaneous bleeding and severe pain in the upper gingiva 2 months before his visit to our hospital, and he had difficulty eating. Although he experienced diarrhea a few times per day, his colitis symptoms remained stable. Clinical examination showed a reddish ulcer with a pebble-like surface, deep fissures, and moderate induration from the right buccal mucosa to the alveolar mucosa (Figure A). Positron emission tomography/computed tomography revealed high uptake in the right maxillary gingiva (Figure B). Initial cytologic examination showed atypical cells containing condensed and moderately enlarged nuclei with irregular nuclear membranes, as well as many leucocytes surrounding these cells. Colonoscopy revealed ulcers, friability, and erythema of the entire colon (Figure C). However, no remarkable change was seen in the ileum. What is the diagnosis of this oral lesion? See the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI.

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Conflicts of interest The authors disclose no conflicts. Funding F.T. is supported by the Japan Society for the Promotion of Science -KAKEN (#26460971). © 2016 by the AGA Institute 0016-5085/$36.00 http://dx.doi.org/10.1053/j.gastro.2016.07.046

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Answer to: Image 5: Pyostomatitis Vegetans

Pyostomatitis vegetans (PV) is a characteristic but rare inflammatory stomatitis that can complicate inflammatory bowel diseases, especially ulcerative colitis.1 The pathogenesis of PV might be associated with cross-reaction of antigens in the bowel and remote organs, including the oral cavity and skin.1,2 When the lesions merge and become necrotic, a characteristic linear “snail track” appearance is noted (Figure A).3 In our case, incisional biopsy revealed a stratified squamous epithelium with pseudoepitheliomatous hyperplasia and an intraepithelial microabscess composed of chronic inflammatory cells (Figure D). Based on the clinical and pathological findings, the patient was diagnosed with PV. PV might persist, and the hyperplastic pathology can mimic a carcinoma; thus, differential diagnosis should be carefully performed. Inflammatory bowel disease treatment is crucial for controlling PV. The use of anti-tumor necrosis factor antibodies to treat PV has not been established; however, a previous case was treated with infliximab.3 Our patient was treated with adalimumab, which lead to mucosal healing of both oral ulcer (Figure E) and colitis (Figure F). The current case highlights the importance of considering PV when persistent ulcers occur in inflammatory bowel disease patients, and it suggests that adalimumab may be a suitable treatment for PV.

References 1. 2. 3.

Shah S, Cotliar J. Images in clinical medicine. Pyostomatitis vegetans. N Engl J Med 2013;368:1918. Ahn BK, Kim SC. Pyodermatitis-pyostomatitis vegetans with circulating autoantibodies to bullous pemphigoid antigen 230. J Am Acad Dermatol 2004;50:785–758. Mijandrusic-Sincic B, Licul V, Gorup L, et al. Pyostomatitis vegetans associated with inflammatory bowel disease–report of two cases. Coll Antropol 2010;34(Suppl 2):279–282.

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