A Case of Gastrointestinal Behçet’s Disease with Actinomycosis of the Lingual Tonsils

Abstract

Actinomycosis is a relatively rare infectious disease that is caused by Actinomyces israelii, which habitually resides as an intraoral normal bacterial flora. The head and neck region is susceptible to actinomycosis infection, but this infection only rarely affects the tongue. On the other hand, Behçet’s disease is a refractory systemic inflammatory disorder characterized by the following four primary symptoms: recurrent oral ulcers, cutaneous symptoms, ocular symptoms, and pubic ulceration. The following three secondary symptoms are also common: intestinal tract lesions, vascular lesions, and central nervous system lesions. Similarly, gastrointestinal Behçet’s disease is defined as a variant in which the primary manifestations are gastrointestinal symptoms. Herein, we report the case of a patient with suspected intestinal Behçet’s disease along with tongue actinomycosis, which was difficult to diagnose due to the rare disease overlap. The patient was an 80-year-old woman who presented with sore throat, and biopsy of the tongue ulcer revealed actinomycosis of the tongue. The sore throat relapsed after the patient received treatment for actinomycosis, and the patient was suspected as having intestinal Behçet’s disease based on the findings of positron emission tomography/computed tomography (PET-CT) and upper-lower gastrointestinal endoscopy. The patient eventually died of pneumonia caused by the long-term steroid therapy. In this case, Behçet’s disease was difficult to diagnose because of the relatively rare affliction of the affected part also by actinomycosis.