Tsugita Nao, Miyagawa Ippei, Nakayamada Shingo, Nawata Aya, Kosaka Shumpei, Ueno Masanobu, Tanaka Yoshiya
The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health, Japan, Kitakyushu, Japan.
Department of Pathology and Oncology, University of Occupational and Environmental Health, Japan, Kitakyushu, Japan.
Mod Rheumatol Case Rep. 2025 Jan 16;9(1):179-183. doi: 10.1093/mrcr/rxae049.
A 55-year-old man was admitted to the hospital with vomiting, diarrhoea, and chest pain. Upon examination, he exhibited signs of increased inflammatory response, acute kidney injury, and thrombocytopenia, leading to a diagnosis of TAFRO syndrome, which was supported by the clinical evidence of generalised lymphadenopathy, pleural effusion, and hepatosplenomegaly. Despite receiving intensive multimodal immunosuppressive therapy, including glucocorticoid pulse therapy (methylprednisolone 1000 mg/day), tocilizumab, and cyclosporine in the intensive care unit, the patient showed minimal response and succumbed to the disease on the seventh day of hospitalisation. Histopathological analysis of the lymph nodes revealed idiopathic multicentric Castleman disease-like features, and Epstein-Barr virus-encoded RNA (EBER) in situ hybridisation identified multiple EBER-positive cells. These findings highlight the elusive pathogenic mechanism of TAFRO syndrome and the potential resistance of some patients to standard treatments such as tocilizumab. The presence of EBER-positive cells in lymph nodes or bone marrow may serve as an indicator of disease severity and treatment resistance. Therefore, histopathological detection of EBER-positive cells may help predict responsiveness to conventional treatments, disease severity, and prognosis in patients with TAFRO syndrome.
一名55岁男性因呕吐、腹泻和胸痛入院。检查时,他表现出炎症反应增强、急性肾损伤和血小板减少的迹象,诊断为TAFRO综合征,全身淋巴结肿大、胸腔积液和肝脾肿大的临床证据支持了这一诊断。尽管在重症监护病房接受了强化多模式免疫抑制治疗,包括糖皮质激素冲击治疗(甲泼尼龙1000毫克/天)、托珠单抗和环孢素,但患者反应甚微,在住院第七天死于该病。淋巴结的组织病理学分析显示具有特发性多中心Castleman病样特征,爱泼斯坦-巴尔病毒编码RNA(EBER)原位杂交鉴定出多个EBER阳性细胞。这些发现突出了TAFRO综合征难以捉摸的致病机制以及一些患者对托珠单抗等标准治疗的潜在耐药性。淋巴结或骨髓中EBER阳性细胞的存在可能作为疾病严重程度和治疗耐药性的指标。因此,EBER阳性细胞的组织病理学检测可能有助于预测TAFRO综合征患者对传统治疗的反应性、疾病严重程度和预后。