Department of Otolaryngology-HNS, Research Institute for Clinical Medicine of Jeonbuk National University, Biomedical Research Institute of Jeonbuk National University Hospital, Jeonbuk, Korea.
Medicine (Baltimore). 2022 Sep 2;101(35):e30175. doi: 10.1097/MD.0000000000030175.
Immunoglobulin G4-related disease (IgG4-RD) has recently been well recognized and Kuttner tumor is known to be a chronic sclerosing sialadenitis, representing the focal manifestation of IgG4-RD, in the submandibular gland (SMG). This study is to evaluate the immunologic features of IgG4-related Kuttner tumor in the SMG. We retrospectively chose 13 patients who were confirmed as having Kuttner tumor by surgical biopsy between May 2012 and January 2019. The fine-needle aspiration cytology, serum antibody levels (anti-Ro antibodies, anti-La antibodies), IgG serum levels (total IgG and IgG4), and immunohistochemical findings for IgG and IgG4-positive plasma cells were reviewed. The cytologic results found that 7 of the 9 cases were reported as chronic sialoadenitis, and the other 3 as benign lymphoproliferative lesion. The serum levels of autoantibodies, Sjögren-syndrome-related antigen A/Ro-Ab and Sjögren-syndrome-related antigen A/Ro-La, showed all normal values of serum level. The serum level of IgG was increased in only 4 among the cases. However, the IgG4 levels were significantly increased in 11 among the cases. In all the patients who received resection of SMG, immunohistochemical findings showed all positive for IgG4-RD, with elevated numbers of IgG and IgG4-positive plasma cells. The evaluation of IgG4 serum level should be very informative for the diagnosis of this tumor before surgery. Fine-needle aspiration cytology with ultrasound guidance are not conclusive in this study. The immunological study including IgG4 serum level should be required for proper diagnosis and treatment, with clinical features of the Kuttner tumor. The level of evidence was IV.
免疫球蛋白 G4 相关疾病（IgG4-RD）最近得到了很好的认识，库特纳瘤被认为是一种慢性硬化性唾液腺炎，是下颌下腺（SMG）中 IgG4-RD 的局灶性表现。本研究旨在评估 SMG 中 IgG4 相关库特纳瘤的免疫学特征。我们回顾性选择了 2012 年 5 月至 2019 年 1 月期间通过手术活检确诊为库特纳瘤的 13 例患者。回顾细针抽吸细胞学、血清抗体水平（抗 Ro 抗体、抗 La 抗体）、IgG 血清水平（总 IgG 和 IgG4）和 IgG 和 IgG4 阳性浆细胞的免疫组织化学发现。细胞学结果发现，9 例中有 7 例报告为慢性唾液腺炎，另 3 例为良性淋巴增生性病变。自身抗体血清水平，干燥综合征相关抗原 A/抗 Ro-Ab 和干燥综合征相关抗原 A/抗 La，均显示正常的血清水平。只有 4 例病例 IgG 血清水平升高。然而，11 例病例的 IgG4 水平显著升高。所有接受 SMG 切除术的患者，免疫组织化学检查均为 IgG4-RD 阳性，IgG 和 IgG4 阳性浆细胞数量增加。在手术前，评估 IgG4 血清水平对于诊断这种肿瘤非常有帮助。在本研究中，超声引导下的细针抽吸细胞学检查并不具有结论性。包括 IgG4 血清水平在内的免疫学研究对于正确诊断和治疗具有临床特征的库特纳瘤是必要的。证据水平为 IV。
