Two cases of pediatric-onset orofacial granulomatosis

Background: Orofacial granulomatosis (OFG) is a general term for rare granulomatous disease that occurs in the oral-facial region without systemic involvement. OFG is characterized by a painless swelling and ulceration of the lips, tongue, and gingiva. It occurs even in relatively young individuals and is often associated with latent Crohn's disease (CD), particularly in children. Case report: We report two cases of OFG in children. Case 1: A 3-year-old boy presented to our hospital with gingival and lip swellings. Biopsy of the gingiva confirmed the formation of a granuloma, and a diagnosis of OFG was made. Oral care and topical application of steroid ointment were administered, and the swelling improved. An endoscopic examination was performed after the perianal lesions were noted, but CD was not diagnosed. However, the patient was observed for possible secondary CD. Case 2: An 8-year-old girl presented with a chief complaint of gingival and lip swelling. Gingival biopsy confirmed granuloma formation, and the diagnosis of OFG was obtained. She was treated with oral tranilast, topical steroid ointment, and oral care, which improved the swelling. Complicated by the gastrointestinal symptoms, the patient was diagnosed with CD using endoscopy. Conclusion: The oral pathophysiologies of OFG and CD are not yet widely understood by dentists or pediatricians. Dentists and oral surgeons were more likely to examine patients with OFG. Therefore, it is necessary to make an appropriate diagnosis, promptly identify the possibility of concomitant or secondary CD, and collaborate with internists and pediatricians for early diagnosis and treatment management.