Sakakibara Ayako, Inagaki Yuichiro, Imaoka Eiki, Ishikawa Eri, Shimada Satoko, Shimada Kazuyuki, Suzuki Yuka, Nakamura Shigeo, Satou Akira, Kohno Kei
Department of Pathology and Laboratory Medicine, Nagoya University Hospital.
Department of Hematology and Oncology, Anjo Kosei Hospital.
J Clin Exp Hematop. 2018 Mar 16;58(1):32-35. doi: 10.3960/jslrt.17037. Epub 2018 Feb 8.
Intravascular large B-cell lymphoma (IVLBCL) is a rare and clinically distinctive entity characterized by the almost exclusive growth of large cells within the lumen of blood vessels in particular capillaries. Reports of this peculiar disease, do not commonly address the PD-L1 expression on IVLBCL tumor cells. Here, we describe a 51-year-old Japanese woman who presented with rapidly progressive cognitive decline and higher brain dysfunction. CT scan and MRI revealed multiple ischemic foci in the cerebral hemispheres, ground-glass opacity in the lungs, and splenomegaly. Random skin biopsy for IVLBCL diagnosis yielded negative results. The patient experienced a rapidly deteriorating clinical course with no treatment, and died from the disease after 3 months of hospitalization. Post-mortem examination revealed systemic intravascular plugging of lymphoma cells, without mass lesions in the central nervous system or in visceral organs such as the lungs, liver, pituitary gland, ovaries, and uterus. The tumor cells were positive for CD10, CD20, BCL2, BCL6, and MUM1, but not other lineage-specific markers. Notably, the tumor cells showed strong PD-L1 expression. Our case was diagnosed as IVLBCL with neoplastic PD-L1 expression. These findings suggest that PD-L1 is associated with immune evasion of IVLBCL and may play a role in the pathogenesis and peculiar biological behavior of this unique disease. Additionally, PD-L1 may represent a possible therapeutic target for immune check-point inhibitors.
血管内大B细胞淋巴瘤(IVLBCL)是一种罕见且具有临床独特性的疾病,其特征是大细胞几乎仅在血管腔尤其是毛细血管内生长。关于这种特殊疾病的报道,通常未涉及IVLBCL肿瘤细胞上的PD-L1表达情况。在此,我们描述一名51岁的日本女性,她出现快速进展的认知衰退和高级脑功能障碍。CT扫描和MRI显示脑半球有多个缺血灶、肺部磨玻璃样混浊以及脾肿大。为诊断IVLBCL进行的随机皮肤活检结果为阴性。该患者未经治疗临床病程迅速恶化,住院3个月后死于该病。尸检显示淋巴瘤细胞全身性血管内阻塞,中枢神经系统或肺、肝、垂体、卵巢及子宫等内脏器官无肿块病变。肿瘤细胞CD10、CD20、BCL2、BCL6和MUM1呈阳性,但其他谱系特异性标志物呈阴性。值得注意的是,肿瘤细胞显示出强烈的PD-L1表达。我们的病例被诊断为具有肿瘤性PD-L1表达的IVLBCL。这些发现表明,PD-L1与IVLBCL的免疫逃逸相关,可能在这种独特疾病的发病机制和特殊生物学行为中起作用。此外,PD-L1可能是免疫检查点抑制剂的一个潜在治疗靶点。