Perianal Kissing Ulcers: Behçet’s Syndrome (with Video)

Excerpt

A 15-year-old male patient with a 2-year history of recurrent oral ulcers presented with recurrent perianal pain for > 1 month. Esophagogastroduodenoscopy revealed three oral ulcers, approximately 0.5–0.8 cm in diameter (Fig. 1a; Video 1). Colonoscopy revealed well-demarcated contralateral ulcers in a kissing configuration in the crissum (Fig. 1b; Video 1). Histopathologic examination of the oral biopsy specimens revealed that the mucosal tissue was covered by mildly atypical squamous epithelial hyperplasia, with more stromal infiltration of lymphocytes and plasma cells infiltration and local ulcer formation (Fig. 1c). Histopathologic examination of the perianal biopsy specimens revealed local chronic inflammation in skin, accompanied with inflammatory exudation and necrosis, small abscesses, and ulcer formation (Fig. 1d). Gomori’s methenamine silver and acid-fast staining of oral and perianal biopsy specimens was negative. Computed tomographic enterography and computed tomography of the chest and abdomen did not reveal overt abnormalities. Pelvic magnetic resonance imaging did not reveal perianal abscesses or anal fistula. Serologic test results were as follows: IgM anti-α-enolase antibody (−), antisacchromyces cerevisia antibody (−), antinuclear antibodies (−), antineutrophil cytoplasimic antibody (−), anti-myeloperoxidase (−), proteinase 3 (−), glomerular basement membrane (−), tolulized red unheated serum test (−), HIV antigen/antibody (−), ferritin: 138.87 ng/ml (reference range, 30.0–406.0), interleukin-6: 8.13 pg/ml (reference range, < 7), and C-reactive protein: 4.50 mg/L (reference range, 0 –6). Human leukocyte antigen-B51 and pathergy tests were positive. The patient was eventually diagnosed with Behçet’s syndrome [1‐3]. All ulcers healed after 1-month treatment with oral prednisone and thalidomide. No recurrence was observed at 4-month follow-up. Here, we present the endoscopic findings of oral ulcers and the rarely observed perianal kissing ulcers in a patient with Behçet’s syndrome.

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