Xiang Yuanyuan, Liu Cuicui, Xue Yuan, Li Shan, Sui Yanling, Li Jifeng, Sun Qinjian, Liu Xiaohui
Department of Neurology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan, China.
Front Neurol. 2020 Sep 11;11:901. doi: 10.3389/fneur.2020.00901. eCollection 2020.
Lymphomatoid granulomatosis (LYG) is an infrequent lymphoproliferative disease that typically involves the lungs, but may also affect the central nervous system (CNS). Isolated CNS involvement is very rare, and its clinicopathological features have not been fully elucidated. Here, we systematically reviewed the English literature through PubMed to collect all relevant case reports and small case series with pathologically confirmed primary CNS-LYG. A total of 29 relevant articles with 40 cases were included in this systemic review. In cases where T cells and B cells were compared, T cells were predominant in 19 (79.2%), and B cells were predominant in 5 (20.8%). The overall infection rate of EBV was 48.1% (13/27), among which the infection rate was 40.9% (9/22) in immunocompetent patients and 80% (4/5) in immunodeficient (HIV-infected) patients. Among the patients who underwent pathological grading, 35.7% (5/14) were at grade I, 42.9% (6/14) were at grade II, and 21.4% were at grade III. In conclusion, primary CNS-LYG is closely related to EBV infection and some cases may be predominantly T-cell phenotype. Surgical resection may be effective for mass-like lesions, although there is still a lack of standard therapeutic regimen. Accurate grading of lesions is essential for treatment selection and prognosis evaluation.
淋巴瘤样肉芽肿病(LYG)是一种罕见的淋巴增殖性疾病,通常累及肺部,但也可能影响中枢神经系统(CNS)。孤立的中枢神经系统受累非常罕见,其临床病理特征尚未完全阐明。在此,我们通过PubMed系统检索英文文献,收集所有经病理证实的原发性中枢神经系统LYG的相关病例报告和小病例系列。本系统评价共纳入29篇相关文章,包含40例病例。在比较T细胞和B细胞的病例中,19例(79.2%)以T细胞为主,5例(20.8%)以B细胞为主。EBV的总体感染率为48.1%(13/27),其中免疫功能正常患者的感染率为40.9%(9/22),免疫缺陷(HIV感染)患者的感染率为80%(4/5)。在接受病理分级的患者中,I级占35.7%(5/14),II级占42.9%(6/14),III级占21.4%。总之,原发性中枢神经系统LYG与EBV感染密切相关,部分病例可能以T细胞表型为主。手术切除对肿块样病变可能有效,尽管仍缺乏标准治疗方案。准确的病变分级对于治疗选择和预后评估至关重要。