Department of Respiratory Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-13 Sugano, Ichikawa, Chiba, 272-0824, Japan.
Department of Pathology and Laboratory Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-13 Sugano, Ichikawa, Chiba, 272-0824, Japan.
BMC Pulm Med. 2020 Apr 25;20(1):104. doi: 10.1186/s12890-020-1150-x.
Immune checkpoint inhibitors (ICIs) are the standard treatment for non-small cell lung cancer. The unique adverse events that can arise after treatment with ICIs are known as immune-related adverse events (irAE). As the number of cases under treatment with ICIs increases, new types of characteristics of irAE have emerged. This case report suggests that IgG4-related pleural disease could occur as an irAE.
A 64-year-old man was diagnosed with pulmonary adenocarcinoma stage IIIB. Following concurrent chemoradiotherapy, durvalumab was administered every two weeks. The patient complained of dyspnea on effort 4 months after the initiation of durvalumab therapy. Chest CT scans showed mild bilateral pleural effusion 4 months after the initiation of durvalumab therapy, and the amount of pleural effusion increased further at 7 months. Durvalumab was thought to be a potential cause of pleural effusion and was withdrawn after 13 courses of administration over 7 months. The level of serum IgG4 was 2750 mg/dL. The levels of IgG4 of the pleural fluids were 2790 mg/dL on the right side and 2890 mg/dL on the left side at 7 months. Microscopic examination of the pleural biopsy revealed lymphoplasmacytic infiltration with storiform fibrosis. Immunohistochemical examinations showed that the number of IgG4-positive cells was > 20/high power field and the percentage of IgG4-positive to IgG-positive plasma cells was > 50%. Oral prednisolone at a dose of 30 mg/day was initiated, and remarkable clinical improvements were achieved. After 4 months of prednisolone therapy, the level of serum IgG4 decreased to 370 mg/dL and chest CT revealed the disappearance of bilateral pleural effusion.
This was a case of IgG4-related pleural disease in a patient with pulmonary adenocarcinoma under durvalumab treatment. To our knowledge, this is the first case report of IgG4-related pleural disease as an irAE. It is important to consider the possibility of IgG4-related pleural disease in cases of pleural effusion during the treatment with ICIs.
免疫检查点抑制剂(ICI)是治疗非小细胞肺癌的标准治疗方法。ICI 治疗后出现的独特不良反应称为免疫相关不良反应(irAE)。随着接受 ICI 治疗的病例数量增加,新的 irAE 特征类型已经出现。本病例报告提示 IgG4 相关胸膜疾病可能作为 irAE 发生。
一名 64 岁男性被诊断为 IIIB 期肺腺癌。同步放化疗后,每两周给予 durvalumab 治疗。durvalumab 治疗开始后 4 个月,患者出现劳力性呼吸困难。胸部 CT 扫描显示 durvalumab 治疗开始后 4 个月时出现双侧轻度胸腔积液,7 个月时胸腔积液量进一步增加。考虑 durvalumab 是胸腔积液的潜在原因,并在 7 个月内给予 13 个疗程后停药。血清 IgG4 水平为 2750mg/dL。7 个月时右侧胸腔液 IgG4 水平为 2790mg/dL,左侧为 2890mg/dL。胸膜活检的显微镜检查显示淋巴浆细胞浸润伴席纹状纤维化。免疫组织化学检查显示 IgG4 阳性细胞数>20/高倍视野,IgG4 阳性浆细胞与 IgG 阳性浆细胞的比例>50%。开始给予 30mg/天的口服泼尼松龙,临床症状显著改善。泼尼松龙治疗 4 个月后,血清 IgG4 水平降至 370mg/dL,胸部 CT 显示双侧胸腔积液消失。
这是一例 durvalumab 治疗肺腺癌患者的 IgG4 相关胸膜疾病。据我们所知,这是首例 IgG4 相关胸膜疾病作为 irAE 的病例报告。在接受 ICI 治疗时,对于胸腔积液患者,应考虑 IgG4 相关胸膜疾病的可能性。