Multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum
Multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum
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DOI：
10.1111/j.1440-1827.2009.02425.x
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发表时间：
2009-09-01
影响因子：
2.2
通讯作者：
Okabe, Hidetoshi
中科院分区：
文献类型：
--
作者：
Ishida, Mitsuaki;Hotta, Machiko;Okabe, Hidetoshi
IgG4相关硬化性疾病被认为是一种独特的临床病理实体。众所周知，这种疾病可以发生在头部和颈部的唾液腺、泪腺和脑垂体。鼻腔是IgG4相关硬化性疾病的一个极其罕见的累及部位。本文报告一例多发性IgG 4相关硬化性病变的上颌窦，腮腺和鼻中隔。一位73岁的日本男性因鼻塞而被诊断为右侧上颌窦和腮腺肿瘤，之后进行了肿瘤切除术。最后一次手术后一年，他注意到鼻中隔肿胀，肿瘤被切除。这三个肿瘤具有相似的组织病理学，如明显的纤维硬化改变伴密集的淋巴浆细胞浸润和偶尔的闭塞性静脉炎。免疫组化显示所有三个病变中有大量IgG 4阳性浆细胞浸润和高比例的IgG 4阳性/IgG阳性浆细胞（> 70%）。诊断为多发性IgG 4相关硬化性病变。本病例提示IgG 4相关硬化性病变可发生于上颌窦和鼻中隔，代表IgG 4相关硬化性疾病谱的扩展。
IgG4-related sclerosing disease is recognized as a distinct clinicopathological entity. It is well known that this disease can occur in the salivary, lacrimal and pituitary glands, in the head and neck region. The nasal cavity is an extremely rare site of involvement of IgG4-related sclerosing disease. Herein is reported a case of multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum. A 73-year-old Japanese man presented with nasal obstruction and tumors of the right maxillary sinus and parotid gland were detected, after which resections of these tumors were performed. One year after the last surgery, he noted swelling of the nasal septum, and the tumor was resected. These three tumors had similar histopathology, such as conspicuous fibrosclerotic changes with dense lymphoplasmacytic infiltration and occasional obliterative phlebitis. Immunohistochemistry indicated abundant IgG4-positive plasma cell infiltration and high ratios of IgG4-positive/IgG-positive plasma cells (> 70%) in all three lesions. The diagnosis of multiple IgG4-related sclerosing lesions was made. The present case suggests that IgG4-related sclerosing lesion can occur in the maxillary sinus and nasal septum, and represents an extension of the spectrum of IgG4-related sclerosing disease.