Behçet’s disease complicated by multiple aseptic abscesses of the liver and spleen

Abstract

Aseptic abscesses are an emergent entity and have been described in inflammatory bowel disease, especially in Crohn’s disease, and in other diseases. However, aseptic abscesses associated with Behçet’s disease are extremely rare. We report a Japanese male diagnosed with an incomplete type of Behçet’s disease who developed multiple aseptic abscesses of the spleen and liver. In 2002, the spleen abscesses were accompanied by paroxysmal oral aphthous ulcers and erythema nodosum. As the patient’s response to antibiotic treatment was inadequate, a splenectomy was performed. Severe inflammatory cell infiltration, largely of polymorphonuclear neutrophils, was observed without evidence of bacterial or fungal growth. Although the patient had no history of ocular symptoms or genital ulcers, a diagnosis of incomplete Behçet’s disease was made according to the Japanese diagnostic criteria because of the presence of paroxysmal arthritis and epididymitis since 2002. In 2005, multiple liver abscesses developed with right hypochondrial pain and seemed to be attributed to Behçet’s disease because the abscesses yielded negative results during a microbiologic investigation and failed to go into remission under antibiotic therapy. Oral prednisone (15 mg/d) was started in May 2006, and the abscesses dramatically disappeared 4 wk after treatment. Although the patient had a relapse of the liver abscesses in association with the tapering of prednisone, the augmentation of prednisone dosage yielded a response. The abscesses of the liver and spleen were strongly suggested to be attributed to Behçet’s disease. Clinician should be aware of the existence of aseptic abscesses as uncommon manifestations of Behçet’s disease.

Keywords: Behçet’s disease; Aseptic abscess; Spleen; Liver; Prednisone

Core tip: We report a Japanese male diagnosed with an incomplete type of Behçet’s disease who developed multiple aseptic abscesses of the spleen and liver. Spleen abscesses developed with paroxysmal oral aphthous ulcers and erythema nodosum. A splenectomy was performed, and severe neutrophil infiltration was observed without evidence of bacterial or fungal growth. Multiple liver abscesses also developed with right hypochondrial pain and seemed to be attributed to Behçet’s disease because the abscesses yielded negative results during a microbiologic investigation. Oral prednisone (15 mg/d) was started, and the abscesses dramatically disappeared. Clinicians should be aware of the existence of aseptic abscesses as uncommon manifestations of Behçet’s disease.