An unusual presentation of Orofacial granulomatosis in a paediatric patient – a case report.

Introduction:
Orofacial granulomatosis (OFG) is an uncommon chronic inflammatory disorder which affects the orofacial region. The main clinical features associated with OFG are non-painful swelling of the lips, angular cheilitis, linear oral ulceration, mucosal tags, and a cobblestone appearance of the buccal mucosa. Histologically, it is associated with noncaseating granulomas and multinucleated Langhans-type giant cells. The aetiology remains unclear and multifactorial factors are implicated thus it is a diagnosis by exclusion.

Presentation:
We present the case of a 16-year-old female patient with known Crohn’s disease and Orofacial granulomatosis who presented with new onset bilateral tongue swelling. On examination, the patient had bilateral lobulated granulomatous changes on her lateral tongue without any other features typical of OFG orofacial disease. These areas were initially resistant to treatment with systemic corticosteroids but subsequently responded to a local delivery topical steroid via a metered inhaler device and the use of a soft mouth guard.

Discussion:
Fissuring of the lateral aspects of the tongue is a known manifestation of OFG, however, to our knowledge granulomatous type changes on the lateral tongue is not a well reported feature of OFG. We discuss the typical features of OFG as described in the literature, report this unusual presentation of OFG and suggest how to approach clinical management.

Reference:
Shetty N, Subramanyam RV, Srikanth G, Solomon MC. Orofacial granulomatosis: A case report and a proposal of a diagnostic algorithm for oral granulomatous lesions. J Oral Maxillofac Pathol. 2021 May-Aug;25(2):346-350. doi: 10.4103/0973-029X.325238. Epub 2021 Aug 31. PMID: 34703131; PMCID: PMC8491324.