Recalcitrant, recurrent aphthous stomatitis successfully treated with adalimumab.

Unidad de Enfermedades Autoinmunes Sistémicas, Hospital Universitario San Cecilio, Granada, Spain.
Journal of the European Academy of Dermatology and Venereology (Impact Factor: 2.69). 06/2008; 23(2):206. DOI: 10.1111/j.1468-3083.2008.02790.x
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    ABSTRACT: Celiac disease is a common autoimmune disease triggered by gluten-containing foods (wheat, barley and rye) in genetically predisposed individuals. We present a patient with celiac disease complicated by severe aphthous stomatitis resulting in impairing swallowing, chewing and speaking. This led to weight loss, psychosocial problems as well as inability to perform her work. A variety of topical and systemic medications used resulted in either no improvement or only partial alleviation of the patient's symptoms. After informed consent, etanercept was initiated and resulted in complete remission of aphthous stomatitis, decrease in arthralgia and fatigue and considerable improvement in her quality of life. The use of newer biological agents for selected and severe manifestations of celiac disease may lead to improved morbidity in these patients, but more studies are needed to determine long-term efficacy as well as safety of these drugs in the mucosal and/or systemic complications of this disease.
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    ABSTRACT: Recurrent aphthous ulcers are the most common inflammatory lesions of the oral mucosa, occurring in up to 10% of the population and even more common in children. The history, morphological characteristics, predilection sides and typical stages of aphthae help to distinguish them from other diseases that may exhibit aphthous-like lesions. Underlying diseases should be excluded. The main goals of therapy are to minimize pain and functional disabilities as well as decrease frequency and severity of recurrences. Topical symptomatic relief is the standard of care for simple cases of recurrent aphthosis. In cases of major aphthosis or systemic involvement, topical therapies are still useful but should be combined with systemic therapy, such as colchicine, pentoxifylline or prednisolone. In case of Adamantiades-Behçet disease, systemic immunomodulatory drugs can inhibit the development of new lesions. This overview summarizes morphological and presentation forms of aphthae, differential diagnoses and evidence-based therapeutic possibilities.
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    ABSTRACT: Abstract Background: Otherwise healthy patients with severe recurrent mucocutaneous aphthous ulcerations (complex aphthosis) may require systemic immunomodulatory therapy. However, a subset of patients remains resistant or intolerant to recommended therapeutic agents. Recently, case reports have described that tumor necrosis factor-alpha (TNF-alpha) inhibitors may induce remission in these patients. Methods: We present data on efficacy and safety of various TNF-alpha inhibitors used as monotherapy in a case series of 18 patients with refractory primary complex aphthosis. Results: Sixteen (89%) patients obtained complete or almost clearance of oro-genital aphthous ulcerations rapidly after onset of therapy either with etanercept, adalimumab, infliximab or golimumab. Duration of treatment ranged between 3 and 77 months. Nine patients (50%) received more than one TNF-alpha inhibitor during the course of treatment. Five (28%) patients experienced side effects that could be related to treatment with TNF-alpha inhibitors. Conclusion: TNF-alpha inhibitors are an effective and safe treatment option for patients with severe complex aphthosis who do not respond sufficiently to standard therapy as recommended by existing guidelines. However, the final position of TNF-alpha inhibitors in the therapeutic armamentarium awaits randomized controlled trials.
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