A 14-year-old, previously healthy, boy presented with a one-month history of persistent swelling of the lower lip. There were no associated symptoms or identifiable inciting events. Physical examination showed soft and painless swelling of the lower lip, particularly on the left side. Two months later, he presented to consultation with permanent lip swelling, weight loss, abdominal pain and vomiting. Initial bloodwork revealed increased inflammatory markers and calprotectin. Gastro-intestinal endoscopies displayed superficial erosions of the colon and esophagus. Mucosal biopsies showed noncaseating transmural granulomas and lip biopsies revealed giant cell granulomas without necrosis. The diagnosis of orofacial granulomatosis associated with Crohn’s disease was confirmed. The patient was treated with azathioprine and mesalamine, with good response.
Orofacial granulomatosis is a rare disorder characterized by facial or lip swelling and can be related to systemic disease. Crohn’s disease presenting as orofacial granulomatosis is very rare in the pediatric population and it can precede the intestinal symptoms of Crohn Disease by months or years. Therefore, it is important to have a low threshold of suspicion and carry out persistent clinical surveillance for the development of additional signs and symptoms.
Key words: Crohn Disease, Orofacial granulomatosis, Pediatrics
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