Double-hit primary central nervous system lymphoma with histogenetically proven bone marrow infiltration: a case report and a review of the literature.

IF 2.7 3区 医学 Q2 CLINICAL NEUROLOGY
Brain Tumor Pathology Pub Date : 2024-10-01 Epub Date: 2024-08-31 DOI:10.1007/s10014-024-00490-z
Koki Onodera, Mitsuaki Shirahata, Reina Mizuno, Masayoshi Fukuoka, Tomonari Suzuki, Tsugumi Satoh, Taku Homma, Naoki Takahashi, Kazuhiko Mishima
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引用次数: 0

Abstract

Double-hit lymphoma (DHL) formerly referred to high-grade B-cell lymphoma with concurrent MYC and BCL2 or BCL6 rearrangements, however, the updated 2022 World Health Organization Classification (5th edition online) excludes those with MYC and BCL 6 rearrangements from the high-grade category. DHL confined to the central nervous system (CNS), known as double-hit primary CNS lymphoma (DH-PCNSL), is rare with poorly understood clinical features. Here, we report a case of a 64-year-old man with multiple brain tumors diagnosed with DH-PCNSL who showed bone marrow (BM) infiltration early in the clinical course. The histological diagnosis was high-grade B-cell lymphoma with MYC and BCL6 rearrangements. Fluorodeoxyglucose positron emission tomography (FDG-PET) revealed no abnormal accumulation except in the CNS. The patient received whole-brain radiotherapy following the failure of high-dose methotrexate. After completion of radiotherapy, the patient developed thrombocytopenia, and BM biopsy showed infiltration of DHL cells, which were not detected by repeated FDG-PET. This is the first report of DH-PCNSL where identical gene rearrangements were confirmed in both the resected CNS tumor and BM tissue. Patients with DH-PCNSL require careful follow-up because they may be at a potential risk of BM infiltration, which may be undetectable by FDG-PET, particularly early in the disease course.

Abstract Image

组织学证实骨髓浸润的双击原发性中枢神经系统淋巴瘤:病例报告和文献综述。
双打淋巴瘤(DHL)以前是指同时伴有MYC和BCL2或BCL6重排的高级别B细胞淋巴瘤,然而,2022年更新的世界卫生组织分类(第5版在线版)将那些伴有MYC和BCL6重排的淋巴瘤排除在高级别类别之外。局限于中枢神经系统(CNS)的DHL被称为双基因突变原发性中枢神经系统淋巴瘤(DH-PCNSL),这种淋巴瘤非常罕见,其临床特征鲜为人知。在此,我们报告了一例 64 岁的男性病例,他患有多发性脑肿瘤,被诊断为 DH-PCNSL,在临床病程早期出现骨髓(BM)浸润。组织学诊断为高级别B细胞淋巴瘤,伴有MYC和BCL6重排。荧光脱氧葡萄糖正电子发射断层扫描(FDG-PET)显示,除中枢神经系统外,其他部位无异常积聚。大剂量甲氨蝶呤治疗失败后,患者接受了全脑放疗。放疗结束后,患者出现血小板减少,骨髓活检显示有DHL细胞浸润,但反复进行FDG-PET检查并未发现。这是首例在切除的中枢神经系统肿瘤和骨髓组织中证实存在相同基因重排的 DH-PCNSL 病例。DH-PCNSL患者需要仔细随访,因为他们可能面临潜在的骨髓浸润风险,FDG-PET可能检测不到这种浸润,尤其是在病程早期。
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来源期刊
Brain Tumor Pathology
Brain Tumor Pathology 医学-病理学
CiteScore
5.40
自引率
9.10%
发文量
30
审稿时长
>12 weeks
期刊介绍: Brain Tumor Pathology is the official journal of the Japan Society of Brain Tumor Pathology. This international journal documents the latest research and topical debate in all clinical and experimental fields relating to brain tumors, especially brain tumor pathology. The journal has been published since 1983 and has been recognized worldwide as a unique journal of high quality. The journal welcomes the submission of manuscripts from any country. Membership in the society is not a prerequisite for submission. The journal publishes original articles, case reports, rapid short communications, instructional lectures, review articles, letters to the editor, and topics.Review articles and Topics may be recommended at the annual meeting of the Japan Society of Brain Tumor Pathology. All contributions should be aimed at promoting international scientific collaboration.
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