Asymptomatic Gut Inflammation in Oro-facial Granulomatosis

Methods: 35 patients with OFG, and no gastrointestinal symptoms were identified and a detailed assessment of the oral cavity performed. Haematological investigation (full blood count, ESR, CRP, vitamin B12, folate, ferritin, SACE and ANA) was undertaken together with a chest radiograph and standard cutaneous patch testing. A biopsy of affected oral mucosa was obtained and an ileocolonoscopy performed under intravenous sedation. A group of patients undergoing ileocolonoscopy for a family history of colorectal cancer acted as the control.

Results: Of the 35 patients (18 female, 17 male) median age was 24 years (range 6-74 years). Median age of onset was 20 years and mean duration 3 years. Lip swelling was commonest presentation but buccal and gingival inflammation were also common. Swelling (91%) and erythema (70%) were commonest macroscopic findings while cobblestoning (49%), fissuring (37%), and ulceration (aphthous-like 15%, linear 12%) were also seen. Haematological and patch test results were not associated with presence of gastrointestinal inflammation. Radiographic examination was normal in all cases. Oral mucosal biopsy demonstrated granulomas in 86% while ileocolonoscopy revealed intestinal abnormalities in 59%, with granulomas demonstrated in 75% of cases. Statistical analysis (Chi squared) revealed a statistically significant relationship between age of onset <30 and GIT involvement, severity of oral involvement and severity of GIT involvement, both p<0.05.

Conclusions: OFG most likely defines a group of conditions in which granulomatous inflammation occurs in the oral cavity, especially the lip. In some cases the inflammation appears limited to the oral cavity. However, in a large proportion, particularly younger patients, the oral inflammation may form part of a generalised, discrete granulomatous enteritis.