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Approach to Orofacial Granulomatosis and Review of Literature

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Approach to Orofacial Granulomatosis and Review of Literature Original Article Approach to orofacial granulomatosis and review of literature A. Javadzadeh 1, A. Pakfetrat 1, F. Falaki 2, S.A. Seyyedi 3 1 Associate Professor, Department of Oral Medicine, School of Dentistry, Mashhad University of Medical Sciences. Mashhad, Iran. 2 Assistant Professor, Department of Oral Medicine, School of Dentistry, Mashhad University of Medical Sciences. Mashhad, Iran. 3 Assistant Professor, Department of Oral Medicine, School of Dentistry, Urmia University of Medical Sciences. Urmia, Iran. Abstract Background and Aim: Orofacial granulomatosis (OFG) comprises a group of diseas- es characterized by non-caseating granulomatous inflammation affecting the soft tis- sues of the oral and maxillofacial region. Wiesenfeld introduced the term orofacial granulomatosis in 1985 for the first time. The precise cause of OFG is unknown; however, some theories have been suggested in- cluding allergy, infection and genetic predisposition. The clinical presentation can be highly variable, making the diagnosis difficult to es- tablish. The aim of this review was to define clinical features, differential diagnosis and treat- ment protocols of OFG. Materials and Methods: All English articles from 1950 to 2010 in Pubmed, InterS- cience, ScienceDirect, Google Scholar databases were searched using the keywords: orofacial granulomatosis, approach, and treatment. Persian articles were also selected from Iran Medex. Results and Conclusion: Because of the relatively nonspecific clinical findings asso- ciated with a variety of granulomatous diseases, the diagnosis of orofacial granulomat- ous often presents a dilemma for the clinician. The most common differential diagno- Corresponding author: sis includes Crohn's disease, sarcoidosis, and infection. However, a variety of other Dr. Seyyedi S.A., Associate conditions may be associated with granuloma formation. Often an extensive clinical, Professor, Department of Oral Medicine, School of Dentistry, microscopic, and laboratory evaluation may be required to reach to definite diagnosis Mashhad University of Medical and proper treatment. Sciences. Mashhad, Iran [email protected] Key Words: Orofacial granulomatosis – Approach – Treatment Received: 26 June 2011 Accepted: 22 Nov 2011 The Journal of Islamic Dental Association of IRAN (JIDA) / Summer 2012 /24 / (2) Introduction treatment and long-term prognosis remains un- Orofacial granulomatosis is an uncommon clini- clear. This article is a comprehensive review of cal and pathological condition. This condition is the existing literature about management, clini- characterized by recurrent and persistent over- cal manifestations, probable etiological factors, growth of oral and maxillofacial soft tissues spe- differential diagnoses and treatment steps of cifically lips and the specific characteristic of orofacial granulomatosis. non-caseous granulomatous inflammation in the History: Orofacial granulomatous lesions unre- absence of detectable systemic diseases such as lated to a certain systemic disease was first re- Crohn’s disease or sarcoidosis. (1,2) The etiolo- ported and described by Melkersson in 1928 as gy of this condition is unclear, therefore its exact an orofacial swelling accompanied with facial 2012; Vol. 24, No. 1 111 The Journal of Islamic Dental Association of IRAN (JIDA) / Summer 2012 /24 / (2) Javadzadeh et. al nerve palsy.(3) Also in 1931, the term–Melkers- in its clinical features, with lips being the most son- Rosenthal Syndrome was defined by Ro- common site of involvement. The frequent ma- senthal to describe the triad of persistent lip and nifestation of the lesion is indicated as recurrent face swelling, facial nerve palsy and fissured labial swellings that have the ability to remain tongue.(4) In 1945, granulomatous lesion of the persistent. (1-9) The swelling is non-tender in lip which was characterized by persistent lip palpation and is initially soft and non-pitting and swelling ( one sign of the Melkersson- Rosenthal later becomes rubbery and firm. Other oral ma- Syndrome) was designated by Meischer as nifestations include: oral ulcers, submucoal Meischer’s cheilitis.(5) Oral granulomatoses swellings, mucosal tags, fissured tongue (lingua were described in accordance with some system- plica), angular cheilitis, gingival overgrowth, ic conditions such as tuberculosis in 1951(6), facial swelling and/or erythema, facial nerve pal- sarcoidosis in 1985(1) and Crohn’s disease in sy, and cervical lymphadenopathy. 2000 (7) But, the term orofacial granulomatosis Lip overgrowth (labial swelling) or oro-facial granulomatosis was first presented Labial overgrowth can involve lower or upper as a scientific term by eldWiesenf in 1985 that lip or both(10). The swelling is often persistent encompasses Melkersson-Rosenthal Syndrome but can also be recurrent, persisting for several cheilitis granulomatosis of Meischer.( fig.1) weeks or months. (11) The swelling may cause Definition: Orofacial granulomatosis is used to enlargement or clefting of the lip(s) (median describe non-caseous granulomatous inflamma- cheilitis) and inflammation and clefting of the tion of the oral and facial region with recurrent corners of the mouth. (angular cheilitis) The and persistent labial swellings in the absence of labial swelling is non-pitting at pressure and any systemic disease. This lesion can be accom- non-tender in palpation and can vary from a soft panied by manifestations such as oral ulcers, to a rubbery consistency based on its persistence. gingival overgrowths, and cobblestone appear- The labial mucosa can be erythematous and have ance of buccal mucosa. (2) In addition, forma- a granular appearance. (9-12) tion of granuloma results in obliteration of lym- Oral ulcers: The three principal types of ulcers phatic vessels, formation of lymphedema, accu- can be encountered in orofacial granulomatosis mulation of interstitial fluid and finally swelling with their most common feature of chronicity. of the lips and other parts of the face. Orofacial In majority of cases, the ulcers are linear and granuloma encompasses conditions previously longitudinal at the depth of the labial or buccal referred to as Melkersson-Rosenthal Syndrome vestibule with exophytic margins with often ery- and Meischer’s cheilitis. Orofacial granuloma is thematous borders. (13-15) an uncommon phenomenon, but diagnosis of The second less common type of ulcers are su- new cases is currently increasing.(1,2) Contro- perficial symmetrical aphthous-like ulcers with versies have recently been arisen about the point well circumscribed borders that can appear in that whether orofacial granuloma is a distinct any part of oral mucosa. clinical entity of a clinical manifestation of cer- The other type of ulcers which are associated tain granulomatous diseases such as Crohn’s with orofacial granulomatosis are ulcers in the disease or sarcoidosis. (8) In addition, other dis- real sense of the word but are described as pus- orders such as profound fungal infections, tuber- tules in anterior part of the gingiva, labial vesti- culosis, allergic angioedema, leprosy, Wegener’s bular mucosa or soft palate. They have the same granuloma, etc. also exist with the same clinical appearance as pyostomatitis vegetans and are not features and are later discussed in differential clinically purulent. In fact the term pustule is diagnosis. used to describe them due to the appearance of Clinical features: Orofacial granuloma I variable intraepithelial leukocytes in their microscopic 112 2012; Vol. 24, No. 2 Javadzadeh et. al Approach to orofacial granulomatosis and review . evaluation. Therefore, this term is not clinically Crohn’s disease, sarcoidosis, etc. Recently, ga- relevant. (16) strointestinal involvement in non-endemic re- Mucosal swellings: Buccal and labial mucosae gions such as southern Europe, Asia and devel- may be swollen producing plications with a cob- oping countries is increasing. There is a possibil- blestone appearance that often involves posterior ity that the prevalence of orofacial granulomato- parts of buccal mucosa. (17,18) sis which has a slight predilection to appear in Mucosal tags: These painless tags of mucosa women increases and manifest primarily in which are often produced at the depth of labial children and young adults. (9,12,19,22) or buccal vestibule, retromolar area or around Etiology and pathogenesis: The exact cause of the chronic ulcerations are orange or red in color. orofacial granulomatosis is currently unknown (17,18) and has been a matter of debate for long. Five Gingival overgrowths: Overgrowths of the free or etiologic factors can be attributed to the orofacial attached gingiva can occur locally or diffusely. granulomatosis. (23-27): They can precede facial or mucosal manifesta- Genetic predisposition tions. The gingiva appears granular with normal Food allergy pink to red in color and rarely ulcerated.(19) Allergy to dental materials Fissured tongue Infection The dorsal surface of the tongue may be fis- Immunologic causes sured. (18) Genetic predisposition: A comprehensive review Facial nerve palsy: Paralysis of the facial motor of the literature does not show evidence to sup- nerve may occur rarely in orofacial granuloma- port genetic causes for orofacial granulomatosis. tosis. This condition can also occur as a result of In a study genetic factors contributed in only formation of granuloma within the nerve trunk. 23% of cases and in another study in 6 out of 42 Facial nerve palsy accompanied with fissured cases. (28) Also, it was reported
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