Abe Takahiro, Sato Tsuyoshi, Tomaru Yasuhisa, Sakata Yasuaki, Kokabu Shoichiro, Hori Naoko, Kobayashi Akio, Yoda Tetsuya
Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Saitama Medical University, Saitama, Japan.
Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009 Oct;108(4):544-50. doi: 10.1016/j.tripleo.2009.06.001.
A new concept of IgG4-related disease characterized by a high serum IgG4 level and tissue infiltration of IgG4-positive plasmacytes that can involve salivary glands has been proposed. In this article, 2 patients with IgG4-related sclerosing sialadenitis involving the submandibular glands are reported. One patient presented with bilateral and painless swelling of the submandibular glands. He had already been treated with systemic prednisolone owing to the occurrence of retrobulbar neuritis. Laboratory examinations showed high serum IgG4 concentrations, and a biopsy of the submandibular gland revealed the infiltration of IgG4-positive plasmacytes. Abdominal computerized tomography demonstrated tumefaction in the tail of the pancreas, thus suggesting localized autoimmune pancreatitis. The other patient also showed bilateral and painless swelling of the submandibular glands, but there was no involvement of any other organs. These patients were effectively treated with corticosteroids, which resulted in a reduction of the swelling of the submandibular gland and increased saliva. When a patient is suspected of having sclerosing sialadenitis, it is important to consider that the patient may have a systemic IgG4-related plasmacytic disease.
一种以血清IgG4水平升高及IgG4阳性浆细胞组织浸润为特征、可累及唾液腺的IgG4相关疾病的新概念已被提出。本文报道了2例累及下颌下腺的IgG4相关硬化性涎腺炎患者。1例患者表现为双侧下颌下腺无痛性肿大。因其发生球后视神经炎,已接受全身性泼尼松龙治疗。实验室检查显示血清IgG4浓度升高,下颌下腺活检显示IgG4阳性浆细胞浸润。腹部计算机断层扫描显示胰腺尾部肿大,提示局限性自身免疫性胰腺炎。另1例患者也表现为双侧下颌下腺无痛性肿大,但无其他器官受累。这些患者接受皮质类固醇治疗有效,下颌下腺肿胀减轻且唾液分泌增加。当怀疑患者患有硬化性涎腺炎时,重要的是要考虑到患者可能患有全身性IgG4相关浆细胞病。