A 15-year-old male patient presented at the Oral Medicine Department of the University Clinical Center with swelling of the upper lip that had persisted for 3 years. The patient claimed that the edema of the lip recurred numerous times each year without causing any pain. There was no history of allergies or systemic disease. The patient's allergic tests for nutritive and inhalational allergens proved negative, in consultation with an allergist.
The clinical examination revealed edema, redness, and fissuring of the upper lip (Figure 1), with unilateral edema and fissures on the buccal mucosa, and gingival hyperplasia in the upper jaw (Figures 2, 3a and b). On palpation, the lip was firm and painless. The gingiva was hyperplastic in both jaws, particularly in the area around the front teeth. Poor oral hygiene was observed. Laboratory investigations revealed that the complete blood count, urinalysis, biochemistry, liver and renal function tests, and thyroid hormones were normal. Consultation with a gastroenterologist ruled out Crohn's disease, and consultation with a pulmonologist excluded tuberculosis and sarcoidosis (PPD test was negative). Following gastroenterological and lung examinations, the diagnosis of an isolated case of orofacial granulomatosis was confirmed. Chest radiography and a series of blood tests were requested to rule out Crohn's disease, tuberculosis, and sarcoidosis. Our patient denied having any gastrointestinal (GI) symptoms but underwent endoscopy and colonoscopy to rule out possibility of CD. His PPD result was negative, however he was referred for a chest X-ray to exclude tuberculosis and sarcoidosis.
On oral panoramic X-rays (Figure 4), periapical alterations in teeth 21 and 22, deep caries in teeth 36, and radix relicta in teeth 46 were clearly discernible. Antibiotics (amoxiclav tablets, 625 mg twice daily) were administered as a premedication before extraction and endodontic treatments.
In consultation with an oral surgeon, we used antibiotics for prophylaxis before tooth extraction and endodontic treatments due to dentoalveolar abscess teeth 36, 46, 21, 22. The antibiotic isn't prescribed to treat OFG, but rather to treat odontogenic infection. Amoxiclav 625 mg twice daily for 1 week was prescribed as prophylaxis.
Endodontic treatment was performed on teeth 21 and 22, and teeth 36 and 46 were extracted to eliminate foci of dentogenic origin. We administered oral prednisone (5 mg twice daily for 10 days) because the edema of the lip and buccal mucosa, fissures, and gingival inflammation were persistent. We injected intralesional steroid injections (40 mg Triamcinolon acetonid) at a frequency of 1 ml per week for 3 weeks to the upper lip because the edema had not entirely subsided. This injection was administered under local anesthesia to avoid the pain associated with the increase of tissue pressure of the edematous lip. After the initial injection, we observed a reduction of the lip size. Six months later, clinical evaluation showed improvement of the lip edema, but with slight cheek swelling, mainly toward the left side, with an asymmetric facial appearance (Figure 5). At the 9-month follow-up, there were no signs of recurrence. No mucosal alterations were present (Figure 6a and b). Non -surgical treatment was started to treat the periodontal inflammation after 9-month of follow-up. The patient provided written informed consent.
A biopsy was performed from a gingival sample. The sample was stained with CK-MNF immunohistochemical stain, which marks epithelial components. Under the squamous epithelium without atypia, dense lymphoplasmacytic inflammatory infiltrate and some multinucleated giant cells could be seen. There was also a proliferation of lymphatic vascular vessels (Figure 7a and b).