Ueki Tomoyuki, Miyake Toru, Narita Mitsuhiro, Kojima Masatsugu, Kaida Sachiko, Iida Hiroya, Shimizu Tomoharu, Tani Masaji
Department of Surgery, Shiga University of Medical Science, Tsukinowa-cho, Seta, Otsu-shi, Shiga, 520-2192, Japan.
Department of Urology, Shiga University of Medical Science, Tsukinowa-cho, Seta, Otsu-shi, Shiga, 520-2192, Japan.
Surg Case Rep. 2020 Aug 3;6(1):197. doi: 10.1186/s40792-020-00964-0.
Immunoglobulin G4-related disease (IgG4-RD) is a novel disease concept of unknown cause that is characterized by abundant infiltration of IgG4-positive cells, mass-forming lesions, and elevated serum IgG4 levels. The infiltration of IgG4-positive plasma cells and lymphocytes causes swelling, inflammation, fibrosis, and obliterative phlebitis in multiple organs. On the other hand, IgG4-RD occurring in the ureters has rarely been reported. To our knowledge, this is the first report of laparoscopic partial ureteral resection for IgG4-related focal retroperitoneal fibrosis in a ureter with suspected colon cancer recurrence.
A 72-year-old man with a history of sigmoid colon cancer visited Shiga University of Medical Science Hospital for regular follow-up in December 2019. Enhanced abdominal computed tomography revealed a mass involving the left ureter. Furthermore, fluorine-18 fluorodeoxyglucose positron emission tomography showed significant accumulation of fluorodeoxyglucose uptake in the same region. Due to the possibility of colon cancer recurrence, a laparoscopic excisional biopsy with partial ureteral resection was performed. Histologically, IgG4-positive plasma cell infiltration exceeding 10 cells per high-power field and a high ratio of IgG4-positive/IgG-positive cells exceeding 40% were observed. The postoperative serum IgG4 level was 384 mg/dL. With the application of these findings to the diagnostic algorithm in the comprehensive diagnostic criteria for IgG4-RD, the mass-forming lesion was diagnosed as definitive IgG4-related focal retroperitoneal fibrosis.
IgG4-RD should be considered in the differential diagnosis of retroperitoneal lesions. Moreover, laparoscopic surgery may be useful for making the diagnosis in difficult-to-biopsy cases.
免疫球蛋白G4相关性疾病(IgG4-RD)是一种病因不明的新型疾病概念,其特征为IgG4阳性细胞大量浸润、形成肿块性病变以及血清IgG4水平升高。IgG4阳性浆细胞和淋巴细胞的浸润会导致多个器官出现肿胀、炎症、纤维化及闭塞性静脉炎。另一方面,IgG4-RD发生于输尿管的情况鲜有报道。据我们所知,这是首例针对疑似结肠癌复发的输尿管IgG4相关性局灶性腹膜后纤维化进行腹腔镜部分输尿管切除术的报告。
一名72岁男性,有乙状结肠癌病史,于2019年12月前往滋贺医科大学医院进行定期随访。增强腹部计算机断层扫描显示左输尿管有一肿块。此外,氟-18氟脱氧葡萄糖正电子发射断层扫描显示同一区域有明显的氟脱氧葡萄糖摄取积聚。由于存在结肠癌复发的可能性,遂进行了腹腔镜切除活检并部分切除输尿管。组织学检查发现,每高倍视野IgG4阳性浆细胞浸润超过10个细胞,且IgG4阳性/I gG阳性细胞的高比例超过40%。术后血清IgG4水平为384mg/dL。将这些发现应用于IgG4-RD综合诊断标准的诊断算法中,该肿块性病变被诊断为确诊的IgG4相关性局灶性腹膜后纤维化。
在腹膜后病变的鉴别诊断中应考虑IgG4-RD。此外,腹腔镜手术可能有助于对难以活检的病例进行诊断。