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Clinical Challenges and Images in GI

2007; Elsevier BV; Volume: 132; Issue: 2 Linguagem: Inglês

10.1053/j.gastro.2007.01.014

1528-0012

Alan C. Moss, Nathaniel S. Treister, Derek K. Marsee, Adam S. Cheifetz,

Neuroendocrine Tumor Research Advances

Question: A 55-year-old woman with active Crohn’s disease presented to clinic with an oral eruption. Six weeks earlier she had presented with a flare of her Crohn’s disease that included an inflammatory mass in her terminal ileum, erythema nodosum, and arthralgias. In addition to her maintenance azathioprine and mesalamine, she was commenced on induction therapy with infliximab 5 mg/kg at 0 and 2 weeks. Three weeks later she complained of generalized tenderness and sensitivity of her lips and painless white lesions throughout her mouth (Figure 1). There was no previous history of similar oral lesions or symptoms. Clinically, her Crohn’s disease had responded very well to infliximab. On examination, she had widespread raised round, spot-like, white lesions on the buccal and labial mucosa and soft palate that in areas had a somewhat reticulated appearance with minimal erythema. There was a single ulceration measuring 3 mm in diameter on the upper left labial mucosa. Swabs for bacterial culture and viral infection were negative. A 4-mm punch biopsy was obtained from the right buccal mucosa. What is the most likely diagnosis? Look on page 829 for the answer and see the Gastroenterology website (http://www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. The biopsy showed acanthosis, parakeratosis, basal cell liquefaction, and a band-like infiltrate of chronic inflammatory cells below the basement membrane with scattered colloid bodies (H&E, original magnification ×100, Figure 2). The clinical and histopathologic appearances were consistent with an oral lichenoid reaction, probably due to her recent infliximab infusions. Given the excellent clinical response of her Crohn’s disease to infliximab, and apparently localized reaction, infliximab infusions were recommenced with close monitoring of the oral lesions. Her lip symptoms were effectively managed with topical tacrolimus therapy, and over the following 4 weeks the lesions began to fade without new lesions. Oral abnormalities in Crohn’s disease are common, but frequently under-recognized by gastroenterologists.1Harty S. Fleming P. Rowland M. Crushell E. McDermott M. Drumm B. Bourke B. A prospective study of the oral manifestations of Crohn’s disease.Clin Gastroenterol Hepatol. 2005; 3: 886-891Abstract Full Text Full Text PDF PubMed Scopus (136) Google Scholar Lichenoid drug reactions have been described in association with many medications, most commonly nonsteroidal anti-inflammatory drugs and angiotensin-converting enzyme inhibitors.2McCartan B.E. McCreary C.E. Oral lichenoid drug eruptions.Oral Dis. 1997; 3: 58-63Crossref PubMed Scopus (90) Google Scholar There is controversy as to whether or not the development of chronic oral lichen planus represents a premalignant condition.3Mignogna M.D. Fedele S. Lo R.L. Lo M.L. Bucci E. Immune activation and chronic inflammation as the cause of malignancy in oral lichen planus: is there any evidence?.Oral Oncol. 2004; 40: 120-130Abstract Full Text Full Text PDF PubMed Scopus (182) Google Scholar The lesions in this case do not have the features of either an anaphylactic or delayed hypersensitivity reaction.4Cheifetz A. Mayer L. Monoclonal antibodies, immunogenicity, and associated infusion reactions.Mt Sinai J Med. 2005; 72: 250-256PubMed Google Scholar Inhibition of tumor necrosis factor-α by infliximab has been associated with impaired clearance of autoreactive T cells, which could explain the pathogenesis in this case; as there are no reports of oral lichenoid reactions to other monoclonal antibodies in the literature.5Kassiotis G. Kollias G. TNF and receptors in organ-specific autoimmune disease: multi-layered functioning mirrored in animal models.J Clin Invest. 2001; 107: 1507-1508Crossref PubMed Scopus (39) Google Scholar In light of the prevalence of oral abnormalities in patients with Crohn’s disease, and the increasing utilization of novel biologic therapies, we recommend that thorough oral examination be a routine component of gastroenterologists’ evaluation of these patients.