9例原发睾丸弥漫大B细胞淋巴瘤临床特点及预后分析
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摘要
目的探讨原发睾丸弥漫大B细胞淋巴瘤(primary testicular diffuse large B-cell lymphoma,PTDLBCL)的临床特征、治疗及预后。方法回顾性分析我院2013年1月至2016年12月确诊的9例PTDLBCL患者的临床资料。结果 9例患者中位发病年龄为64岁(50~72岁),以右侧睾丸受累为主;Ann Arbor分期以早期为主,其中Ⅰ期6例,Ⅲ期1例,Ⅳ期2例;细胞来源以非生发中心来源为主,非生发中心B细胞来源7例,生发中心B细胞来源2例;肿瘤增殖指数Ki-67偏高,Ki-67中位数为80%。治疗以手术加术后辅助化疗为主,其中单纯手术治疗1例,手术联合CHOP方案为主的化疗8例。获完全缓解6例,部分缓解2例,疾病进展1例。随访时间5~34个月,存活6例,死亡3例。结论原发睾丸弥漫大B细胞淋巴瘤是一组罕见的疾病,复发风险高,一线治疗方案推荐患侧睾丸根治性切除术后联合R-CHOP方案化疗,同时对对侧睾丸及受累野进行放疗及预防性甲氨蝶呤鞘内化疗。
Objective To analyze the clinical characteristics,outcomes and treatment strategies of a case series of patients with primary testicular diffuse large B-cell lymphoma(PTDLBCL). Methods Various clinical parameters were analyzed in 9 patients with PTDLBCL diagnosed from January 2013 to September 2016 in our hospital. Results Median age at diagnosis was 64 years(range,50-72). Cases involved mainly the right testicle,and cells of origin were non-GCB subtype(7 patients) or GCB subtype(2 patients).Median rate of Ki-67 expression was 80%.One patient underwent surgery only,while seven had surgery followed by chemotherapy.Six patients showed complete response;two,partial response;and one,progression.Six patients were alive,corresponding to follow-up ranging from 5 to 34 months. Conclusion PTDLBCL has high recurrence risk. We suggest first-line treatment involving 6-8 cycles of anthracycline-based chemotherapy and rituximab treatment,central nervous system prophylaxis and contralateral testicular radiation after surgery.
Objective To analyze the clinical characteristics,outcomes and treatment strategies of a case series of patients with primary testicular diffuse large B-cell lymphoma(PTDLBCL). Methods Various clinical parameters were analyzed in 9 patients with PTDLBCL diagnosed from January 2013 to September 2016 in our hospital. Results Median age at diagnosis was 64 years(range,50-72). Cases involved mainly the right testicle,and cells of origin were non-GCB subtype(7 patients) or GCB subtype(2 patients).Median rate of Ki-67 expression was 80%.One patient underwent surgery only,while seven had surgery followed by chemotherapy.Six patients showed complete response;two,partial response;and one,progression.Six patients were alive,corresponding to follow-up ranging from 5 to 34 months. Conclusion PTDLBCL has high recurrence risk. We suggest first-line treatment involving 6-8 cycles of anthracycline-based chemotherapy and rituximab treatment,central nervous system prophylaxis and contralateral testicular radiation after surgery.
引文
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[15]Brouwer CL,Wiesendanger EM,van der Hulst PC,et al.Scrotal irradiation in primary testicular lymphoma:review of the literature and in silico planning comparative study[J].Int J Radiat Oncol Biol Phys,2013,85(2):298-308.
[16]Kraan W,van Keimpema M,Horlings HM,et al.High prevalence of oncogenic MYD88 and CD79B mutations in primary testicular diffuse large B-cell lymphoma[J].Leukemia,2014,28(3):719-720.
[17]Chen J,Xu-Monette ZY,Deng L,et al.Dysregulated CXCR4 expression promotes lymphoma cell survival and independently predicts disease progression in germinal center B-cell-like diffuse large Bcell lymphoma[J].Oncotarget,2015,6(8):5597-5614.
[18]Beider K,Ribakovsky E,Abraham M,et al.Targeting the CD20 and CXCR4 pathways in non-hodgkin lymphoma with rituximab and high-affinity CXCR4 antagonist BKT140[J].Clin Cancer Res,2013,19(13):3495-3507.
[19]Escoubet-Lozach L,Lin IL,Jensen-Pergakes K,et al.Pomalidomide and lenalidomide induce p21 WAF-1 expression in both lymphoma and multiple myeloma through a LSD1-mediated epigenetic mechanism[J].Cancer Res,2009,69(18):7347-7356.
[20]Li Z,Qiu Y,Personett D,et al.Pomalidomide shows significant therapeutic activity against CNS lymphoma with a major impact on the tumor microenvironment in murine models[J].PLo S One,2013,8(8):e71754.
[2]Vitolo U,Ferreri AJ,Zucca E.Primary testicular lymphoma[J].Crit Rev Oncol Hematol,2008,65(2):183-189.
[3]Cheah CY,Wirth A,Seymour JF.Primary testicular lymphoma[J].Blood,2014,123(4):486-493.
[4]Deng L,Xu-Monette ZY,Loghavi S,et al.Primary testicular diffuse large B-cell lymphoma displays distinct clinical and biological features for treatment failure in rituximab era:a report from the International PTL Consortium[J].Leukemia,2016,30(2):361-372.
[5]Herráez-Albendea MM,Jarilla-Fernández MC.Primary testicular lymphoma[J].Actas Urol Esp,2016,40(1):68.
[6]Mazloom A,Fowler N,Medeiros LJ,et al.Outcome of patients with diffuse large B-cell lymphoma of the testis by era of treatment:the M.D.Anderson Cancer Center experience[J].Leuk Lymphoma,2010,51(7):1217-1224.
[7]Tohda S.Overview of lymphoid neoplasms in the fourth edition of the WHO classification[J].Rinsho Byori,2012,60(6):560-564.
[8]Lu TX,Miao Y,Wu JZ,et al.The distinct clinical features and prognosis of the CD10(+)MUM1(+)and CD10(-)Bcl6(-)MUM1(-)diffuse large B-cell lymphoma[J].Sci Rep,2016,6:20465.
[9]Cheson BD,Pfistner B,Juweid ME,et al.Revised response criteria for malignant lymphoma[J].J Clin Oncol,2007,25(5):579-586.
[10]Cao B,Ji DM,Zhou XY,et al.A clinical analysis of primary testicular diffuse large B-cell lymphoma in China[J].Hematology,2011,16(5):291-297.
[11]Read JA,Koff JL,Nastoupil LJ,et al.Evaluating cell-of-origin subtype methods for predicting diffuse large B-cell lymphoma survival:a meta-analysis of gene expression profiling and immunohistochemistry algorithms[J].Clin Lymphoma Myeloma Leuk,2014,14(6):460-467.
[12]Cheah CY,Oki Y,Westin JR,et al.A clinician′s guide to double hit lymphomas[J].Br J Haematol,2015,168(6):784-795.
[13]Horwitz SM,Zelenetz AD,Gordon LI,et al.NCCN Guidelines Insights:Non-Hodgkin's Lymphomas,Version 3.2016[J].J Natl Compre Cancer Netw,2016,14(9):1067-1079.
[14]Richie JP.Re:First-line treatment for primary testicular diffuse large B-cell lymphoma with rituximab-CHOP,CNS prophylaxis,and contralateral testis irradiation:final results of an international phaseⅡtrial[J].J Urol,2012,188(1):115-116.
[15]Brouwer CL,Wiesendanger EM,van der Hulst PC,et al.Scrotal irradiation in primary testicular lymphoma:review of the literature and in silico planning comparative study[J].Int J Radiat Oncol Biol Phys,2013,85(2):298-308.
[16]Kraan W,van Keimpema M,Horlings HM,et al.High prevalence of oncogenic MYD88 and CD79B mutations in primary testicular diffuse large B-cell lymphoma[J].Leukemia,2014,28(3):719-720.
[17]Chen J,Xu-Monette ZY,Deng L,et al.Dysregulated CXCR4 expression promotes lymphoma cell survival and independently predicts disease progression in germinal center B-cell-like diffuse large Bcell lymphoma[J].Oncotarget,2015,6(8):5597-5614.
[18]Beider K,Ribakovsky E,Abraham M,et al.Targeting the CD20 and CXCR4 pathways in non-hodgkin lymphoma with rituximab and high-affinity CXCR4 antagonist BKT140[J].Clin Cancer Res,2013,19(13):3495-3507.
[19]Escoubet-Lozach L,Lin IL,Jensen-Pergakes K,et al.Pomalidomide and lenalidomide induce p21 WAF-1 expression in both lymphoma and multiple myeloma through a LSD1-mediated epigenetic mechanism[J].Cancer Res,2009,69(18):7347-7356.
[20]Li Z,Qiu Y,Personett D,et al.Pomalidomide shows significant therapeutic activity against CNS lymphoma with a major impact on the tumor microenvironment in murine models[J].PLo S One,2013,8(8):e71754.