Sialolithiasis in the Duct of Submandibular Gland: A Case Report in Patient with Epidermolysis Bullosa

Abstract

To describe the options of treatment to remove a sialolith associated with the submandibular gland duct in a patient with epidermolysis bullosa (EB). Treatment of patients with EB is very complex and involves a multidisciplinary team. This condition is characterized by a spectrum of blistering and mechanical fragility of the skin. One main feature of this disease is the esophageal constriction and possible constriction to the submandibular duct. This alteration may induce the formation of calculi in this duct, which is called sialolith. Once the sialolith obliterates the trajectory of the duct this will lead to a sialolithiasis. The calculi have to be removed. Seventeen years old female patient with dystrophic EB developed a sialolith at the submandibular duct. She has a limited mouth opening and her tongue was collapsed with mouth floor. The first choice of treatment was the lithotripsy, once this procedure is less invasive and a surgical remove could worse the collapsed tongue. She was with acute pain and with a great augmentation in the submandibular area. Once the patient was debilitated and has difficult to swallow she invariably needed to be hospitalized in order to receive intravenous medication. During the hospitalization the sialolith could be seen through the opening of the duct and the calculi was removed with local anesthesia. The treatment of sialolithiasis usually does not present major challenges, nevertheless if the sialolithiasis is associated with EB, the treatment became an extremely challenge. In this particular case the option of treatment was the less invasive. This case report has an enormous clinical relevance once there is no protocol to treat patients with EB and buccal diseases.