Progressive multifocal leukoencephalopathy (PML) remains a life-threatening central nervous system infection in immunocompromised patients. Although outcomes have improved in cases that immune reconstitution is feasible with anti-retroviral therapy (ART) in HIV + patients or natalizumab removal in those with multiple sclerosis, in individuals with hematological malignancies, the prognosis is usually dismal. Anti-viral treatments have been largely ineffective, but immunotherapy-based approaches with checkpoint inhibitors and adoptive virus-specific T cells’ transfer are currently explored in clinical trials. PML has not been described as a cause of encephalopathy after CAR T therapy. We report the first case of PML 7 months after lymphodepleting chemotherapy with fludarabine/cyclophosphamide and anti-CD19-directed CAR T therapy in a patient with relapsed diffuse large B-cell lymphoma who relapsed fast after a previous autologous hematopoietic stem cell transplant. She remains alive 12 months after diagnosis with stabilization of her symptoms with a combination of therapies targeting viral replication and immunotherapy.

在免疫功能低下的患者中，进行性多灶性白质脑病（PML）仍然是威胁生命的中枢神经系统感染。尽管在HIV +患者中使用抗逆转录病毒疗法（ART）进行免疫重建是可行的，在多发性硬化症患者中使用那他珠单抗去除免疫重建是可行的，但对于血液系统恶性肿瘤患者，预后通常是令人沮丧的。抗病毒治疗在很大程度上无效，但是目前正在临床试验中探索基于免疫疗法的检查点抑制剂和过继性病毒特异性T细胞转移的方法。没有将PML描述为CAR T治疗后脑病的病因。我们报告了氟达拉滨/环磷酰胺和抗CD19导向的CAR T治疗淋巴结清扫化疗后7个月的第一例PML，该患者复发性弥漫性大B细胞淋巴瘤在先前的自体造血干细胞移植后迅速复发。诊断后12个月，她通过结合病毒复制和免疫疗法的多种疗法使症状稳定，从而得以存活。