BILATERAL SUBMANDIBULAR KÜTTNER TUMOR AS INITIAL MANIFESTATION OF PRIMARY SJOGREN SYNDROME: AN UNUSUAL CASE REPORT

• Published in: Oral surgery, oral medicine, oral pathology and oral radiology Vol. 134; no. 3; pp. e108 - e109
• Main Authors: De Almeida Lima Borba Lopes, Amanda, De Souza Vieira, Gustavo, Silva, Arley, Milagres, Adrianna, Rozza-De-Menezes, Rafaela Elvira, Cunha, Karin Soares Gonçalves, Conde, Danielle Castex
• Format: Journal Article
• Language: English
• Published: Elsevier Inc 01-09-2022
• ISSN: 2212-4403; 2212-4411
• DOI: 10.1016/j.oooo.2022.01.155

Sjögren's syndrome is a chronic autoimmune disease, and Küttner tumor is a benign inflammatory condition. Both affect the salivary glands and are rarely associated. A 48-year-old Black woman complained of bilateral submandibular salivary gland enlargement, xerostomia, and xerophthalmia for approximately 7 months. Sialometry revealed severe hyposalivation. An ultrasound examination performed 4 months after the onset of symptoms showed a solid hypoechogenic nodule of 20 mm in the left submandibular salivary gland. An incisional biopsy demonstrated a chronic sclerosing sialadenitis (Küttner tumor) with only 10% positivity for IgG4+ plasma cells, ruling out the diagnosis of IgG4-related disease. Serologic exam for anti-SSA/Ro antibody was highly reactive (200.0 U/mL). Labial salivary glands were excised, and the histopathologic exam revealed a focal lymphocytic sialadenitis, confirming the diagnosis of Sjögren's syndrome. Prednisone (40 mg) and methotrexate (15 mg) were administered for 1 month, with symptom improvement and clinical reduction of the volume of the Küttner tumor.