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115:Orofacial Granulomatosis Treated with Combination of Intralesional Steroids and Hydroxychloroquine[AAOM2020}
EP35997
Poster Title: 115:Orofacial Granulomatosis Treated with Combination of Intralesional Steroids and Hydroxychloroquine[AAOM2020}
Submitted on 29 Mar 2021
Author(s): Mohammed Bindakhil, D.D.S, M.S.1, Eric T Stoopler, D.M.D.2, Faizan Alawi, D.D.S.3, Thomas P Sollecito, D.M.D.2
Affiliations: 1Department of Oral Biology and Diagnostic Sciences, Division of Oral Medicine, The Dental Collage of Georgia, Augusta University 2Department of Oral Medicine, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania. 3Division of Oral and Maxillofacial Pathology, Hospital of the University of Pennsylvania
This poster was presented at 2021 American Academy of Oral Medicine Virtual Conference
Poster Views: 81
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Poster Information
Abstract: Background: Orofacial granulomatosis (OFG) is an uncommon granulomatous inflammatory condition characterized by recurrent or persistent swelling of the orofacial soft tissues, oral ulcerations, gingivitis, and/or cheilitis. Histologically, OFG demonstrates noncaseating subepithelial granulomas and inflammatory cell infiltrates. Multiple treatment options, including intralesional steroid injections, antimicrobial therapy and immunosuppressants, have been used with variable results.

Case summary: A 39-year-old female presented for evaluation of a persistent upper-lip swelling for the past two years. The patient was previously evaluated by several providers and completed a long-term antihistamine treatment consisting of 10 mg daily of cetirizine and 600 mg daily of ranitidine without benefit. Medical history was significant for postural orthostatic tachycardia syndrome and allergic rhinitis. Medication consisted of Loestrin 30mg daily for contraception. Review of systems revealed diaphoresis, shortness of breath with exercise, heart palpitations, and intermittent dry cough. Clinical examination revealed significant swelling of the upper lip with crusted epithelium and a localized slightly raised lesion on the maxillary anterior gingiva. Differential diagnosis included OFG, sarcoidosis, angioedema, and Crohn's disease. Chest X-ray was unremarkable. Assessment of antineutrophil cytoplasmic antibodies, angiotensin-converting enzyme, and C1 esterase inhibitor were within normal limits. Incisional biopsy of gingival tissue revealed numerous granulomas composed of epithelioid histiocytes admixed with multinucleated giant cells and a diffuse infiltrate of lymphocytes and plasma cells. Esophagogastroduodenoscopy and colonoscopy revealed no evidence of inflammatory bowel disease. Cumulative findings were consistent with the diagnosis of OFG. Intralesional injections of 0.3 mL of triamcinolone (10/mL) to the upper lip were performed every eight weeks and 400mg hydroxychloroquine daily was prescribed. The patient experienced significant improvement with this treatment combination.

Conclusion: Systemic granulomatous disorders, including sarcoidosis and Crohn's disease, may resemble OFG. It is necessary to exclude conditions with similar clinical or histomorphologic features. Treatment of OFG can be challenging due to limited evidence of therapeutic benefit with many documented treatments. Combination therapy may be necessary for effective management of this condition.
Summary: Orofacial granulomatosis (OFG) is an uncommon granulomatous inflammatory condition characterized by recurrent or persistent swelling of the orofacial soft tissues, oral ulcerations, gingivitis, and/or cheilitis. Histologically, OFG demonstrates noncaseating subepithelial granulomas and inflammatory cell infiltrates.

Ask the author questions about this poster:Bindakhil@hotmail.com
References: 1. Miest R, Bruce A, Rogers RS 3rd.. Orofacial granulomatosis. Clin Dermatol. 2016;34(4): 505–513.
2. Gale G, Östman S, Saalman R, Telemo E, Jontell M, Hasséus B. Immunophenotype in orofacial granulomatosis with and without Crohn's disease. Med Oral Patol Oral Cir
Bucal. 2014;19(6): 584–591.
3. Alawi F. An update on granulomatous diseases of the oral tissues. Dent Clin North Am. 2013; 57(4): 657–671.
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