Diagnostic and Therapeutic Value of Sialendoscopy in a Patient with Kussmaul Disease: A Case Report

Kussmaul disease is characterized by recurrent pain and swelling in the major salivary glands that arise from obstruction of salivary ducts by mucofibrinous plugs. In this case report, a 15-year-old girl presented with complaints of pain and recurrent swelling in both parotid regions exacerbated by food intake, which had been persisting for 2 years. History revealed allergic asthma and allergic rhinitis. Head and neck examination revealed diffuse swelling and tenderness in the both parotid glands, mucofibrinous plug drainage from the opening of the bilateral Stensen's ducts, diffuse nasal polyposis, and bilateral otitis media with effusion. The serum eosinophil and the serum immunoglobulin E levels were high while the erythrocyte sedimentation rate and C-reactive protein levels were normal. Computed tomography of the head and neck revealed diffuse ductal ectasia with the enlarged Stensen's duct in the parotid gland and diffuse increased density in all the paranasal sinuses. Yellowish mucofibrinous plugs were seen within the Stensen's duct on sialendoscopy. Histopathological examination of the mucofibrinous plugs demonstrated Charcot-Leyden crystals with profuse eosinophils. In conclusion, mucofibrinous plugs and the enlarged salivary ducts on sialendoscopy may be a sign of Kussmaul disease diagnosis. Sialendoscopy may be a useful diagnostic method and therapeutic approach along with other relevant treatment methods for Kussmaul disease.