A Case of Painful Oral Ulceration

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Purpose: 35-year-old man with history of left sided ulcerative colitis for 2 years, presented to GI clinic with complaints of burning oral pain. A week prior to the onset of oral pain, he noticed flu-like symptoms and began to notice painful ulcers in the buccal aspect of cheeks that progressively spread to the gums and the under surface of the tongue. Patient had decreased oral intake due to the pain. He denied joint pains or skin rashes. He did not have a prior history of oral ulceration. He was having 1-2 well formed bowel movements per day with no blood or mucus. He had been well controlled on 4.8 grams of mesalamine. His vitals were stable and his exam was unremarkable other than multiple, small, superficial oral ulcers with whitish base that did not have the typical appearance of aphthous ulcers. Patient was started on viscous lidocaine for symptomatic relief. There was concern for acute necrotizing gingivitis and patient was started on a one week course of amoxicillin with no improvement in his symptoms. Trial of diflucan and nystatin resulted in no improvement in his symptoms. Patient underwent biopsy that revealed intraepithelial abscess with many eosinophils and neutrophils consistent with pyostomatitis vegetans. Patient was started on steroids with complete resolution of his symptoms. Pyostomatitis vegetans is an extremely rare oral condition associated with inflammatory bowel disease, particularly ulcerative colitis. In most cases, bowel disease precedes the onset of oral lesions by months or years. A peripheral eosinophilia has been observed in most reported cases. Pyostomatitis vegetans is characterized by erythematous, thickened oral mucosa with multiple pustules and superficial erosions. Intraepithelial and subepithelial eosinophilic miliary abscesses are characteristically seen on histology. The pathogenesis is yet unknown although immunological and microbial factors have been suggested as possible etiological factors. Management of pyostomatitis vegetans consists mainly of immunosuppression using topical or systemic corticosteroids with medical and/or surgical treatment of any underlying inflammatory bowel disease.