Abstract
Recurrent oral ulcer (ROU) is a common condition that significantly impacts quality of life. It is often related to systemic diseases, such as Behçet’s disease (BD), Crohn’s disease, and ulcerative colitis. Treatment of ROU depends on its severity: from topical agents for mild cases to systemic agents, such as corticosteroids, azathioprine, or other immunosuppressants for severe cases. Recently, good results have been reported with infliximab in refractory ROU. However, the optimal dosage and treatment duration have not been determined and the cost and potential side effects should be considered. We report on four patients who received a single-dose infliximab for refractory ROU. Two patients had refractory ROU with no underlying disease; one of them had soft palate perforation accompanied by severe oral ulcers. The two other patients had ROU of BD without major organ involvement. All patients received a single infusion of infliximab and an additional infusion was given on demand in one patient. Infliximab showed a rapid, good response in three patients and was also effective in improving the acute inflammation in the perforation of the soft palate, which had been resistant to conventional therapies. These effects diminished over a few weeks, but the ROU were tolerable and it was not necessary to increase steroids or add another medicine for about 1 year. We suggest that a single infusion of infliximab can be considered for refractory ROU.
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Introduction
Recurrent oral ulcers (ROU) is a common condition that may be related to systemic diseases, such as celiac disease, Crohn’s disease (CD), ulcerative colitis (UC), Behçet’s disease (BD), and Sweet syndrome . It is often termed “recurrent aphthous stomatitis (RAS)” in the absence of a systemic disease . The onset of ROU seems to peak in teenagers, becoming less frequent with advancing age. It is essential to consider a possible systemic cause, especially when adult patients suddenly develop ROU .
Treatments for ROU depend on how they affect the patient’s quality of life, because they typically do not lead to significant morbidity or mortality . No curative treatment for ROU is yet available . According to the consensus approach for RAS treatment, the first step is to identify underlying precipitating factors and avoid them . However, if they cannot be avoided and RAS makes the quality of life worse, topical steroids are recommended as the first-line treatment . In refractory ROU that do not respond to topical agents, immunosuppressants, including systemic steroids, azathioprine, levamisole, colchicine, pentoxifylline, and dapsone, can be tried .
Recently, anti-tumor necrosis factor-alpha agents (anti-TNF-α) have been shown to produce good responses in ROU . However, there are no clinical guidelines on the dosage or treatment duration of anti-TNF-α for ROU. We describe three cases treated with just a infliximab single infusion and one case with a single and additional infusions of infliximab on demand for refractory ROU and a review of the literature.