Case of Suspected Sialodochitis Fibrinosa (Kussmaul’s Disease)

Here we report a case of Kussmaul’s disease, or sialodochitis fibrinosa. This rare disease is characterized by recurrent swelling of the salivary glands, which then discharge clots of fibrin into the oral cavity. An 80-year-old man with a history of allergic rhinitis visited our department with the chief complaint of pain in the bilateral parotid gland area on eating. An initial examination revealed mild swelling and tenderness in this region, and indurations could be felt around the bilateral parotid papillae. Pressure on the parotid glands induced discharge of gelatinous plugs from the parotid papillae. No pus was discharged, and there were no palpable hard objects. Panoramic X-ray showed no obvious focus of dental infection, and there was no calcification in the parotid gland region. Magnetic resonance imaging revealed segmental dilatation of the main ducts of both parotid ducts, with no signs of displacement due to sialoliths or tumors, or of abnormal saliva leakage. Two courses of antibiotic therapy resulted in no improvement. During treatment, gelatinous plugs (fibrin clots) obstructing the left parotid duct were dislodged by massage, which prevented further blockage by encouraging salivary outflow. The obstruction persisted in the right parotid duct, however. Therefore, the distal portion of the right parotid duct was partially resected and the opening into the mouth enlarged, which, in combination with massage, prevented further obstruction. The pain and swelling of the parotid gland and discharge of gelatinous plugs improved, with no further recurrence at 12 months postoperatively. This case is presented along with a review of the relevant literature.