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Orofacial Granulomatosis: a Review

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Orofacial Granulomatosis: a Review DOI Number: 10.5958/0974-1283.2019.00181.6 Orofacial Granulomatosis: A Review Saloni Banga1, M K Sunil2, Hina Padaliya1 1Intern, 2Professor and Head of Department, Department of Oral Medicine & Radiology, Teerthanker Mahaveer Dental College & Research Center Moradabad Abstract Orofacial Granulomatosis (OFG) is an uncommon clinicopathological entity characterized clinically by the presence of persistent enlargement of the soft tissues of the oral and maxillofacial region and histologically by non-caseating and non-necrotizing granulomatous inflammation. The term ‘orofacial granulomatosis’ has been introduced to denote the group of various disorders, including Melkersson-Rosenthal syndrome and granulomatous cheilitis and has been noted to be associated with Crohn’s disease, Sarcoidosis and infectious diseases such as Tuberculosis. Although, various etiological agents such as dental materials, food additives and microbial agents have been recommended in the disease process. Treatment of orofacial granulomatosis is by corticosteroids but it’s not so efficient. It is more important to identify the pathogen first to specify the appropriate treatment line. Keywords— orofacial granulomatosis, Melkersson Rosenthal syndrome, crohn,s disease. Introduction Definition and its associated to other granulomatous disorders Orofacial granulomatosis term was introduced by Wiesenfeld in 19851. Granulomatosis is any condition Orofacial granulomatosis is an uncommon disorder1 characterized by the formation of multiple nodules or but is increasingly recognized. Orofacial granulomatosis granulomas in soft tissues. It encompasses Melkersson (as defined by Wiesenfeld in 1985) is the specific Rosenthal syndrome and Cheilitis granulomatosa1. histological finding of granulomas in mucosal or skin The true prevalence of this disease is unknown but is biopsies taken from the mouth or face in the absence suggested to be 0.8% (Mahler and Kiesewetter, 1996)3. of a recognised systemic condition known to cause it is principally a condition of children and young granulomas. adults, common in both genders2 with slight female Orofacial granulomatosis includes a group of predilection. It is also associated with oral ulceration, disorders showing chronic, non-caseating granulomatous gingival overgrowth and a cobblestone appearance of lesions involving the perioral tissue of face and oral the buccal mucosa 4. The diagnosis can be confirmed mucosa and whose diagnosis is based on exclusion by histopathological identification of non-caseating of possible systemic diseases such as Tuberculosis, granulomas 5. The precise etiology of Orofacial Sarcoidosis4,8,9. It can cause significant cosmetic and granulomatosis is unknown. Genetics, allergies (food, functional problems but can be prevented if diagnosed dental materials), microbial agents or immunology were early and treated promptly.10 Most common diseases suggested as potential causative agents6,7. of orofacial granulomatosis involving head and neck region8 are Corresponding author: 1. Melkersson Rosenthal syndrome Dr M.k. Sunil 2. Sarcoidosis Professor and Head, Dept of Oral Medicine and Radiology, Teerthankar Mahaveer Dental College and 3. Crohn’s disease Research Center, Moradabad Email [email protected] 4. Midline lethal granuloma 248 Medico-legal Update, July-December 2019, Vol.19, No. 2 5. Wegener’s granulomatosis CROHN’S DISEASE (CD) MELKERSSON ROSENTHAL SYNDROME It encompasses a group of disorder with specific (MRS) clinical ad pathological features, characterized by focal asymmetric, trans mural and occasionally granulomatous It is a neuro-muco-cutaneous disorder involving inflammation primarily affecting the gastrointestinal both the intermittent orofacial innervations and tract.36 It has now been described as a pan-electric disease mucocutaneous tissues in a pathosis of complex origin that can affect any part of the gastrointestinal tract, from characterized by recurrent oedema, facial palsies mouth to anus22. Idiopathic inflammatory bowel disease and nerve dysfunctions frequently associated with affecting the small intestine was first reported in 1932 plicated tongue11,12. However, many patients did not by Crohn and his workers. The first report of Crohn’s manifest all signs of this triad. Monosymptomatic or disease affecting mouth was made by Dudeney in 1969, oligosymptomatic are two forms in which only one or describing a tag on the buccal mucosa of a 36 year old two features of the triad are present commonly.13,14 patient. Melkersson reported a case describing facial Young patients with recurrent painful intraoral palsy and orofacial oedema in 1928 with symptoms ulcerative lesions with no signs of systemic disease, apart of facial palsy and orofacial swelling. Subsequently, from weight loss are affected. Clinical manifestations of Rosenthal in 1932 described the triad of persistent lip non-specific and specific lesions include23 or facial swelling, recurrent facial oedema, swelling and fissured tongue.15 Thus, the term Melkersson Rosenthal • Cobblestone appearance of buccal mucosa. syndrome was derived by Hornstein et al and Worsae • Epithelial tags and folds. et al and reported forms of Melkersson Rosenthal syndrome as monosymptomatic/ oligosymptomatic • Full thickening and swelling of the gingiva. which was described by Miescher in 1945 as Cheilitis granulomatosis.16 • Linear apthous ulceration. It is manifested as orofacial oedema which affects • Redness of attached gingiva extending to the face, lips, gingiva, buccal mucosa or tongue.14 Upper mucosal margins. lip swelling begins as the first symptom and follows • Persistent enlarged rubbery lip swelling.23 lower lip swelling involving one or both cheeks.17 The enlarged lip appears cracked and fissured with reddish MIDLINE LETHAL GRANULOMA brown discoloration and scaling.18 It is the ulcerative process that occurs in the nose SARCOIDOSIS characterized by epithelium and cartilage loss with crusting resulting in loss of nasal structure, support and Sarcoidosis is described as a multisystem ultimately causing cosmetic and functional deformity.11 granulomatous disease of unknown origin characterized Midline destructive lesions of the face were first reported by the formation of uniform, discrete, compact, non- in 1897. Stewart et al reported 10 cases of a chronic caseating epithelioid granulomas19. It is also known as destructive midfacial process in 1922 and named as Boeck sarcoid and Besnier-Boeck-Schaumann disease20. Midline lethal granuloma.24 The common presenting Jonathan Hutchinson, an English surgeon- dermatologist, symptoms include chronic rhinosinusitis refractory to reported the first case of sarcoidosis in 1875, but this term treatment and midline lesion destroy normal sinonasal sarcoidosis was introduced later by Boeck in 1899, which anatomy.24 in Greek means, “flesh-like” condition. The etiology is unknown. Most common feature is pulmonary infiltration WEGENER GRANULOMATOSIS like dry cough, dyspnoea and Lofgren syndrome.25 On the palate and buccal mucosa, the lesions are bleb like, It is a condition associated with generalised vasculitis containing a clear yellowish fluid or as solid nodules21. and was reported by Heinz Klinger and Frederick 25 It also appears that Sarcoidosis may produce diffuse Wegener in 1936 . The classical tissue abnormality destruction of bone. in all organs affected by Wegener granulomatosis is Medico-legal Update, July-December 2019, Vol.19, No. 2 249 inflammation with granuloma formation against a causation of orofacial granulomatosis follows notation of 26 non-specific inflammatory background. Hyperplastic infective agents associated with chronic granulomatous granular gingivitis or “strawberry gingivitis” is a rare conditions such as Crohn’s disease, sarcoidosis and manifestation of Wegener’s granulomatosis, but it is tuberculosis. Several authors have inspected the probable nearly pathognomic for this multisystem autoimmune role of microbial factors in initiating the immune response 26 vasculitis . of orofacial granulomatosis, involving M.Tuberculosis, Saccharomyces cerevisiae, M.Paratuberculosis, Candida AETIOPATHOGENESIS albicans and Streptococcus Mutans. Although a number of possible causative agents have been associated to orofacial granulomatosis21, the CLINICAL FEATURES actual etiopathogenesis remains unknown. Labial enlargement and sometimes oral ulcers HEREDITARY AND GENETIC PREDISPOSITION are primarily the clinical manifestations of orofacial granulomatosis, but numerous other features can also be According to the available literature, there is no seen. adequate data that shows that orofacial granulomatosis has a definite genetic background.4,27 An association LABIAL ENLARGEMENT between orofacial granulomatosis and human leukocyte It involves upper or lower lip or both10,32. The antigen (HLA) has been seen and the two studies swelling is often persistent but can be recurrent also- present do not depict a strong link between HLA and each episode taking weeks to months33. Non-tender in 27 pathogenesis of orofacial granulomatosis. palpation, non-pitting at pressure and in consistency FOOD ALLERGY AND ALLERGY TO DENTAL it may vary from soft to rubbery, labial mucosa can 10,32,33 MATERIALS be erythematous and have granular appearance . Affected individuals may develop a lip licking habit Various food substances and food additives24 have that gives rise to consequential cheilitis with swelling, been suggested to be either the cause or the predisposing redness and drying of perioral skin34. even
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