A rare case report of orofacial granulomatosis in a pediatric patient

Abstract

Orofacial granulomatosis comprises a group of diseases characterized by noncaseating granulomatous inflammation affecting the soft tissues of the oral and maxillofacial region. The most common clinical presentation is persistent swelling of one or both lips. It is important to establish the diagnosis accurately because this condition is sometimes a manifestation of Crohn's disease or sarcoidosis. This article describes a case of orofacial granulomatosis.

Clinicopathological diagnosis of orofacial granulomatosis

“…Local and systemic conditions characterized by granulamatous inflammation must be excluded by appropriate clinical and laboratory investigations. [ 6 ] OFG characteristically presents with lip swelling along with affecting the gingivae, buccal mucosa, floor of the mouth, and a number of sites in the oral cavity. [ 7 ] However, the clinical presentation can be highly variable, making the diagnosis difficult to establish.…”

“…For example, intraoral involvement may take the form of hypertrophy, erythema, or nonspecific erosions involving the gingiva, oral mucosa, or tongue. [ 6 8 ]…”

“…Upon microscopic identification of granulomatous inflammation, special stains are used to rule out deep fungal infectons [periodic acid schiff stain (PAS)] or specific bacterial infections (Ziehl-Neelsen, Gram). [ 6 ] Blood tests, radiology, and endoscopy are other tools used to differentiate OFG from Crohn's disease, sarcoidosis, tuberculosis, and foreign body reactions. [ 5 ] A diagnosis of idiopathic OFG is made on the basis of negative results of a thorough investigation.…”

“…However, rare spontaneous remission is possible. [ 6 15 ] First line treatment involves the use of local or systemic corticosteroids or both. [ 6 ] Other alternative treatment agents have been reported in the literature including hydroxychloroquine, methotrexate, azathioprine, clofazimine, metronidazole, minocycline, thalidomide, dapsone, danazol, and TNF-α blocking agents such as infliximab and adalimumab.…”

Orofacial granulomatosis affecting lip and gingiva in a 15-year-old patient: A rare case report

“…Intra-lesional injections have been proven a better therapy over the systemic corticosteroids therapy. [5] Higher volume intra-lesional 3–10 ml of triamcinolone (10 mg/ml) injection has been used in patients with OFG. Before injection, nerve block anesthesia enables adequate volume and repeated injections to be given painlessly.…”