发生甲氨蝶呤相关周围神经毒性的母细胞性浆细胞样树突细胞肿瘤患儿1例临床分析并文献复习  

Clinical analysis of a case with methotrexate related peripheral neurotoxicity in children with blastic plasmacytoid dendritic cell tumor and literature review

作　　者：刘文慧 吴涛 刘洋 Liu Wenhui;Wu Tao;Liu Yang(Department of Hematology,the 940th Hospital of Joint Logistics Support Force of Chinese People′s Liberation Amy,Lanzhou 730050,Gansu Province,China)

机构地区：[1]中国人民解放军联勤保障部队第九四〇医院血液科,兰州730050

出　　处：《国际输血及血液学杂志》2024年第6期512-519,共8页International Journal of Blood Transfusion and Hematology

基　　金：甘肃省创新基地和人才计划(21JR7RA015);甘肃省重点研发计划(22YF7FA106);联勤保障部队第九四〇医院血液病医学研究中心项目(2021yxky078)。

摘　　要：目的探讨发生甲氨蝶呤相关周围神经毒性的1例母细胞性浆细胞样树突细胞肿瘤(BPDCN)患儿的临床特征及诊疗方法,并进行相关文献复习。方法选择2023年6月20日中国人民解放军联勤保障部队第九四〇医院血液科收治的1例12岁男性BPDCN患儿(患者1)为研究对象。采用回顾性分析方法,收集其临床病例资料,包括临床特征,实验室检查(外周血和骨髓细胞形态学、免疫组织化学等)及细胞遗传学和分子生物学(染色体核型、基因突变等)相关检查结果,以及诊断、治疗及预后资料。根据患者临床症状及相关检查结果对其进行诊断和治疗。以"母细胞性浆细胞样树突细胞肿瘤""甲氨蝶呤""周围神经毒性""儿童""blastic plasmacytoid dendritic cell neoplasm""methotrexate""peripheral neurotoxicity""child"为中、英文关键词,在中国知网数据库、万方数据知识服务平台及PubMed数据库中,检索BPDCN患儿应用甲氨蝶呤后诱发神经毒性的相关临床研究文献,并进行总结。文献检索时间为上述数据库建库至2024年5月20日。本研究符合2013年修订的《世界医学协会赫尔辛基宣言》要求,并与受试者监护人签订临床研究知情同意书。结果①患者1因"间断咳嗽、咳痰1个月,胸闷、气短、发热1 d"于2023年5月1日于当地医院门诊就诊,其胸部CT检查提示肺部感染,外周血细胞形态结果示幼稚细胞比例为35%。为行进一步诊疗,于2023年6月20日转入本院。②入院后骨髓细胞形态学检查结果示幼稚细胞比例为42%。免疫组织化学结果示,58.3%的细胞群符合BPDCN表型,表达CD4、CD123、CD56和人类白细胞抗原(HLA)-DR,部分表达CD45、CD7、CD38,不表达CD34、CD117、CD2、cCD3、CD10、CD13、CD14、CD15、CD19、CD20、CD33、CD64和髓过氧化物酶(MPO)。染色体核型为46,XY,del(1)(p36p33),del(6)(q21q24),der(9)t(9;?)(q32;?),add(12)(q24)。BPDCN相关融合基因呈阴性。具有临床意义的变异为Objective To investigate the clinical features,diagnosis,and treatment of methotrexate related peripheral neurotoxicity in children with blastic plasmacytoid dendritic cell neoplasm(BPDCN)and to review relevant literature.Methods On June 20,2023,one case of 12-year-old male patient(patient 1)with BPDCN was admitted to the Department of Hematology at the 940th Hospital of the Joint Logistics Support Force of the Chinese People′s Liberation Army.A retrospective analysis was conducted to collect clinical data,including clinical features,laboratory tests(peripheral blood and bone marrow cell morphology,immunophenotyping,etc.),cytogenetic and molecular biological results(chromosomal karyotype,gene mutations,etc.),as well as diagnostic,therapeutic,and prognostic information.The patient was diagnosed and treated based on clinical symptoms and relevant test results.The China National Knowledge Infrastructure database,Wanfang data knowledge Service Platform,and PubMed database were searched using the keywords"blastic plasmacytoid dendritic cell neoplasm""methotrexate""peripheral neurotoxicity"and"child"in both Chinese and English.Relevant clinical research literature on BPDCN and methotrexate related peripheral neurotoxicity in children was retrieved and summarized.The search period ranged from the establishment of each database to May 20,2024.Procedures followed in this study were in accordance with the requirements of the World Medical Association Declaration of Helsinki revised in 2013.Informed consent of clinical research was signed with guardian of the subject.Results①Patient 1 was admitted to a local hospital on May 1,2023,due to"intermittent cough and expectoration for one month,along with chest tightness,shortness of breath and fever for one day".Chest CT indicated a pulmonary infection,and the result of peripheral blood cell morphology showed that proportion of immature cells was 35%.For further diagnosis and treatment,he was transferred to our hospital at June 20,2023.②After admission,bone marrow cell morp

关 键 词：母细胞性浆细胞样树突细胞肿瘤 甲氨蝶呤 神经毒性 周围神经病 儿童 

分 类 号：R733.1[医药卫生—肿瘤]