Recalcitrant, Recurrent Aphthous Stomatitis Treated With Etanercept

Abstract

A 50-year-old woman presented with a 24-year history of recurrent aphthous stomatitis. Nine to 12 new lesions usually appeared weekly, resolving in approximately 2 weeks. She experienced associated pain, dysphagia, and swelling of the lips and tongue with a subjective pain severity of 9 (scale, 1-10). Symptoms were exacerbated before menstrual periods and were unchanged with use of oral contraceptives and after menopause. There was no association with food or oral trauma, but she reported flares with emotional stress. She denied associated fever, genital ulcers, skin lesions, or ocular complaints. On physical examination, there were small erosions ( 5 mm) on the left lateral part of the tongue, gingival mucosa, and nonkeratinized labial mucosa, surrounded by erythematous halos (Figure 1). A complete blood cell count, levels of serum urea nitrogen, creatinine, electrolytes, vitamins B1, B2, B6, B12, folate, zinc, ferritin, serum iron, thyrotropin, and antinuclear antibodies, and herpes simplex virus culture werewithin normal limits or negative. Patch testing for sensitivity to mercury chloride amalgams was also negative. A biopsy specimen showed a mucosal ulceration with acute and chronic inflammation. Frequent tapered courses of oral prednisone resulted in improvement of pain and healing of ulcers. Intramuscular and topical corticosteroids, viscous lidocaine, acyclovir, oral erythromycin, doxycycline, clotrimazole troches, and mouthwashes consisting of diphenhydramine hydrochloride, tetracycline hydrochloride, and prednisone provided only minimal and transient improvement of symptoms. Dapsone was started November 1995 but discontinued because of angioedema. Colchicine was started March 1996 but not tolerated because of diarrhea. Minocycline, doxycycline, and acyclovir were not effective. Thalidomide, 50 to 100 mg daily, was started in November 1998, with significant improvement in the patient’s symptoms. However, thalidomide was discontinued because of a sensory peripheral neuropathy in her toes. She started treatment with gabapentin, which resulted in an improvement in her neuropathic symptoms. On discontinuation of thalidomide, she experienced flares consisting of 9 to 12 new erosions every 2 weeks with a subjective pain severity of 8, once again requiring systemic corticosteroids.