原发性腮腺淋巴瘤的临床病理特点及预后分析
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摘要
目的:探讨原发性腮腺淋巴瘤的临床病理特点及影响其预后的因素。方法:收集2006—2016年于北京大学口腔医院口腔颌面外科就诊并诊断为原发性腮腺淋巴瘤患者的相关临床资料,结合复查随访资料,回顾性总结其临床病理特点及患者预后,分析影响预后的相关因素。结果:研究共纳入41例患者,中位年龄57岁(8个月至91岁),男∶女=1∶2. 15,女性多见。40例(97. 1%)为非霍奇金淋巴瘤(non-Hodgkin lymphoma,NHL),其中结外边缘区黏膜相关淋巴组织淋巴瘤(extranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue,MALT) 15例,弥漫性大B细胞淋巴瘤(diffuse large B cell lymphoma,DLBCL) 14例,滤泡型淋巴瘤(follicular lymphoma,FL) 4例,其他类型少见,霍奇金淋巴瘤(Hodgkin lymphoma,HL)仅占1例。Ann ArborⅠE～ⅡE期37例(90. 2%),ⅢE～ⅣE期4例(9. 8%)。7例(17. 1%)淋巴瘤由干燥综合征恶变而来,均为MALT淋巴瘤。平均病期为20. 7月,78%的患者以腮腺区缓慢生长的无痛性肿块为主要症状。治疗方法包括局部治疗及系统治疗,不同治疗方案均可取得较好疗效。全部患者均经5～149个月随访,其中9例(21. 9%)死亡,2年总生存率为84. 5%,5年总生存率为81. 3%。单因素分析显示有无肿物加速生长史(P=0. 005)和肿物有无包膜(P=0. 011)为总生存率的影响因素,多因素分析显示肿物有无包膜(P=0. 041)影响患者的总生存期。结论:腮腺淋巴瘤绝大多数为B细胞型NHL,MALT淋巴瘤与DLBCL最为常见,大部分患者表现为缓慢生长的腮腺区局限性肿块,临床上需与腮腺良性肿瘤相鉴别。干燥综合征与MALT淋巴瘤的发病密切相关。原发性腮腺淋巴瘤患者预后比其他恶性肿瘤相对较好,肿物无包膜提示预后较差。
Objective: To investigate the clinicopathological features and prognostic factors in lymphoma of parotid origin. Methods: Clinicopathological data of the patients with parotid lymphoma who were initially diagnosed in Peking University Hospital of Stomatology from 2006 to 2016 were collected and analyzed retrospectively. The patients were followed-up for 5 to 149 months with a median period of 45 months,and the factors influencing the prognosis were evaluated. Results: A total of 41 patients with primary parotid lymphoma were included in this retrospective study. The rate of male to female was1 ∶ 2. 15. The median age was 57 years(ranging from 8 months to 91 years). According to WHO classification,40 cases(97. 1%) were diagnosed as non-Hodgkin lymphoma(NHL),including 15 cases of extranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue(MALT),14 cases of diffuse large B cell lymphoma(DLBCL) as well as 4 cases of follicular lymphoma,while other subtypes of NHL were rare. Only one case was diagnosed as Hodgkin lymphoma. Regarding the clinical staging at the initial diagnosis,37 patients(90. 2%) were diagnosed as stage ⅠE or ⅡE of the disease,while 4 patients(9. 8%) as stage ⅢE or stage ⅣE. Seven patients(17. 1%) had a history of Sj9 gren syndrome(SS),all of whom were MALT lymphoma. The mean ill duration of the 41 patients were 20. 7 months.Thirty-two patients(78%) presented a slowly growing painless mass in the parotid gland. Treatment options included localized therapy and systemic therapy,all of whom had good curative effect. Nine patients(21. 9%) died during the follow-up period. The overall survival rates of 2-year and 5-year were84. 5% and 81. 3% respectively. The univariate analysis demonstrated statistically significant differences for accelerated growth of tumor(P = 0. 005),and presence of tumor capsule(P = 0. 011). The multiunivariate analysis demonstrated statistically significant differences for presence of tumor capsule(P =0. 041). Conclusion: A large majority of primary parotid lymphoma were NHL,among which MALT lymphoma and DLBCL were common subtypes. Most patients presented an indolent mass in parotid gland,which should be distinguished from the parotid benign tumors. SS is closely related to the pathogenesis of MALT lymphoma. The prognosis is better than that of other malignant parotid tumors. Absence of tumor capsule may predict a poor prognosis in patients with parotid lymphoma.
Objective: To investigate the clinicopathological features and prognostic factors in lymphoma of parotid origin. Methods: Clinicopathological data of the patients with parotid lymphoma who were initially diagnosed in Peking University Hospital of Stomatology from 2006 to 2016 were collected and analyzed retrospectively. The patients were followed-up for 5 to 149 months with a median period of 45 months,and the factors influencing the prognosis were evaluated. Results: A total of 41 patients with primary parotid lymphoma were included in this retrospective study. The rate of male to female was1 ∶ 2. 15. The median age was 57 years(ranging from 8 months to 91 years). According to WHO classification,40 cases(97. 1%) were diagnosed as non-Hodgkin lymphoma(NHL),including 15 cases of extranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue(MALT),14 cases of diffuse large B cell lymphoma(DLBCL) as well as 4 cases of follicular lymphoma,while other subtypes of NHL were rare. Only one case was diagnosed as Hodgkin lymphoma. Regarding the clinical staging at the initial diagnosis,37 patients(90. 2%) were diagnosed as stage ⅠE or ⅡE of the disease,while 4 patients(9. 8%) as stage ⅢE or stage ⅣE. Seven patients(17. 1%) had a history of Sj9 gren syndrome(SS),all of whom were MALT lymphoma. The mean ill duration of the 41 patients were 20. 7 months.Thirty-two patients(78%) presented a slowly growing painless mass in the parotid gland. Treatment options included localized therapy and systemic therapy,all of whom had good curative effect. Nine patients(21. 9%) died during the follow-up period. The overall survival rates of 2-year and 5-year were84. 5% and 81. 3% respectively. The univariate analysis demonstrated statistically significant differences for accelerated growth of tumor(P = 0. 005),and presence of tumor capsule(P = 0. 011). The multiunivariate analysis demonstrated statistically significant differences for presence of tumor capsule(P =0. 041). Conclusion: A large majority of primary parotid lymphoma were NHL,among which MALT lymphoma and DLBCL were common subtypes. Most patients presented an indolent mass in parotid gland,which should be distinguished from the parotid benign tumors. SS is closely related to the pathogenesis of MALT lymphoma. The prognosis is better than that of other malignant parotid tumors. Absence of tumor capsule may predict a poor prognosis in patients with parotid lymphoma.
引文
[1]陈万青,郑荣寿,张思维,等. 2013年中国恶性肿瘤发病和死亡分析[J].中国肿瘤,2017,26(1):1-7.
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[13] Ambrosetti A,Zanotti R,Pattaro C,et al. Most cases of primary salivary mucosa-associated lymphoid tissue lymphoma are associated either with Sj9gren syndrome or hepatitis C virus infection[J]. Br J Haematol,2004,126(1):43-49.
[14] Dong L,Chen Y,Masaki Y,et al. Possible mechanisms of lymphoma development in Sj9gren’s syndrome[J]. Curr Immunol Rev,2013,9(1):13-22.
[15] Zhang W,Feng S,Yan S,et al. Incidence of malignancy in primary Sj9gren’s syndrome in a Chinese cohort[J]. Rheumatology(Oxford),2010,49(3):571-577.
[16] Theander E,Henriksson G,Ljungberg O,et al. Lymphoma and other malignancies in primary Sj9gren’s syndrome:a cohort study on cancer incidence and lymphoma predictors[J]. Ann Rheum Dis,2006,65(6):796-803.
[17]卢松鹤,闫志敏,魏明洁,等.结节型舍格伦综合征与恶性淋巴瘤关系初探[J].中华口腔医学杂志,2012,47(4):208-213.
[18] Peveling-Oberhag J,Arcaini L,Bankov K,et al. The antilymphoma activity of antiviral therapy in HCV-associated B-cell non-Hodgkin lymphomas:a meta-analysis[J]. J Viral Hepat,2016,23(7):536-544.
[19] Arcaini L,Rossi D,Lucioni M,et al. The NOTCH pathway is recurrently mutated in diffuse large B-cell lymphoma associated with hepatitis C virus infection[J]. Haematologica,2015,100(2):246-252.
[20] Brito-Zerón P,Gheitasi H,Retamozo S,et al. How hepatitis C virus modifies the immunological profile of Sj9gren syndrome:analysis of 783 patients[J]. Arthritis Res,2015,17(1):1-9.
[21] Raderer M,Kiesewetter B,Ferreri AJ. Clinicopathologic characteristics and treatment of marginal zone lymphoma of mucosa-associated lymphoid tissue(MALT lymphoma)[J]. CA Cancer J Clin,2016,66(2):153-171.
[22] Swerdlow SH,Campo E,Pileri SA,et al. The 2016 revision of the World Health Organization classification of lymphoid neoplasms[J]. Blood,2016,127(20):2375-2390.
[23] Thielker J,Grosheva M,Ihrler S,et al. Contemporary management of benign and malignant parotid tumors[J]. Front Surg,2018,39(5):1-17.
[24] Gudmundsson JK,Ajan A,Abtahi J. The accuracy of fine-needle aspiration cytology for diagnosis of parotid gland masses:a clinicopathological study of 114 patients[J]. J Appl Oral Sci,2016,24(6):561-567.
[25] Orita Y,Sato Y,Kimura N,et al. Characteristic ultrasound features of mucosa-associated lymphoid tissue lymphoma of the salivary and thyroid gland[J]. Acta Otolaryngol,2014,134(1):93-99.
[26]高润涛,耿雪霏. 8例腮腺恶性淋巴瘤诊疗体会[J].口腔医学研究,2017,33(8):870-873.
[27] Olszewski AJ,Desai A. Radiation therapy administration and survival in stageⅠ/Ⅱextranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue[J]. Int J Radiat Oncol Biol Phys,2014,88(3):642-649.
[28] Jackson AE,Mian M,Kalpadakis C,et al. Extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue of the salivary glands:a multicenter,international experience of 248 patients(IELSG 41)[J]. Oncologist,2015,20(10):1149-1153.
[29] Salles G,Barrett M,Foa R,et al. Rituximab in B-cell hematologic malignancies:a review of 20 years of clinical experience[J]. Adv Ther,2017,34(10):2232-2273.
[30]傅志英,朱军,宋玉琴,等. 525例弥漫大B细胞淋巴瘤预后影响因素分析[J].北京大学学报(医学版),2014,46(3):405-411.
[31]杨小芸,沈丽达,龙庭凤,等. 1 326例非霍奇金淋巴瘤临床病理特点分析[J].中华肿瘤防治杂志,2016,23(9):605-609.
[2]李小秋,李甘地,高子芬,等.中国淋巴瘤亚型分布:国内多中心性病例10 002例分析[J].诊断学理论与实践,2012,11(2):111-115.
[3] Urquhart A,Berg R. Hodgkin’s and non-Hodgkin’s lymphoma of the head and neck[J]. Laryngoscope,2001,111(9):1565-1569.
[4] Etemad-Moghadam S,Tirgary F,Keshavarz S,et al. Head and neck non-Hodgkin’s lymphoma:a 20-year demographic study of381 cases[J]. Int J Oral Max Surg,2010,39(9):869-872.
[5] Swerdlow SH,Campo E,Harris NL,et al. WHO classification of tumours of haematopoietic and lymphoid tissues[M]. 4th ed.Iyon:International Agency for Research on Cancer,2017.
[6] Skarin AT,Dorfman DM. Non-Hodgkin’s lymphomas:Current classification and management[J]. CA Cancer J Clin,1997,47(6):351-372.
[7] Gleeson MJ,Bennett MH,Cawson RA. Lymphomas of salivary glands[J]. Cancer,1986,58(3):699-704.
[8] El-Naggar AK,Chan JKC,Grandis JR,et al. WHO classification of head and neck tumors[M]. 4th ed. Lyon:International Agency for Research on Cancer,2017:276-280.
[9] Feinstein AJ,Ciarleglio MM,Cong X,et al. Parotid gland lymphoma:prognostic analysis of 2140 patients[J]. Laryngoscope,2013,123(5):1199-1203.
[10] Vazquez A,Khan MN,Sanghvi S,et al. Extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue of the salivary glands:a population-based study from 1994 to 2009[J]. Head Neck,2015,37(1):18-22.
[11] Isaacson P,Wright DH. Malignant lymphoma of mucosa-associated lymphoid tissue. A distinctive type of B-cell lymphoma[J].Cancer,1983,52(8):1410-1416.
[12]徐鹏程,周心一,周晨,等.腮腺黏膜相关淋巴组织淋巴瘤的临床特点分析[J].临床耳鼻咽喉头颈外科杂志,2017(1):61-64.
[13] Ambrosetti A,Zanotti R,Pattaro C,et al. Most cases of primary salivary mucosa-associated lymphoid tissue lymphoma are associated either with Sj9gren syndrome or hepatitis C virus infection[J]. Br J Haematol,2004,126(1):43-49.
[14] Dong L,Chen Y,Masaki Y,et al. Possible mechanisms of lymphoma development in Sj9gren’s syndrome[J]. Curr Immunol Rev,2013,9(1):13-22.
[15] Zhang W,Feng S,Yan S,et al. Incidence of malignancy in primary Sj9gren’s syndrome in a Chinese cohort[J]. Rheumatology(Oxford),2010,49(3):571-577.
[16] Theander E,Henriksson G,Ljungberg O,et al. Lymphoma and other malignancies in primary Sj9gren’s syndrome:a cohort study on cancer incidence and lymphoma predictors[J]. Ann Rheum Dis,2006,65(6):796-803.
[17]卢松鹤,闫志敏,魏明洁,等.结节型舍格伦综合征与恶性淋巴瘤关系初探[J].中华口腔医学杂志,2012,47(4):208-213.
[18] Peveling-Oberhag J,Arcaini L,Bankov K,et al. The antilymphoma activity of antiviral therapy in HCV-associated B-cell non-Hodgkin lymphomas:a meta-analysis[J]. J Viral Hepat,2016,23(7):536-544.
[19] Arcaini L,Rossi D,Lucioni M,et al. The NOTCH pathway is recurrently mutated in diffuse large B-cell lymphoma associated with hepatitis C virus infection[J]. Haematologica,2015,100(2):246-252.
[20] Brito-Zerón P,Gheitasi H,Retamozo S,et al. How hepatitis C virus modifies the immunological profile of Sj9gren syndrome:analysis of 783 patients[J]. Arthritis Res,2015,17(1):1-9.
[21] Raderer M,Kiesewetter B,Ferreri AJ. Clinicopathologic characteristics and treatment of marginal zone lymphoma of mucosa-associated lymphoid tissue(MALT lymphoma)[J]. CA Cancer J Clin,2016,66(2):153-171.
[22] Swerdlow SH,Campo E,Pileri SA,et al. The 2016 revision of the World Health Organization classification of lymphoid neoplasms[J]. Blood,2016,127(20):2375-2390.
[23] Thielker J,Grosheva M,Ihrler S,et al. Contemporary management of benign and malignant parotid tumors[J]. Front Surg,2018,39(5):1-17.
[24] Gudmundsson JK,Ajan A,Abtahi J. The accuracy of fine-needle aspiration cytology for diagnosis of parotid gland masses:a clinicopathological study of 114 patients[J]. J Appl Oral Sci,2016,24(6):561-567.
[25] Orita Y,Sato Y,Kimura N,et al. Characteristic ultrasound features of mucosa-associated lymphoid tissue lymphoma of the salivary and thyroid gland[J]. Acta Otolaryngol,2014,134(1):93-99.
[26]高润涛,耿雪霏. 8例腮腺恶性淋巴瘤诊疗体会[J].口腔医学研究,2017,33(8):870-873.
[27] Olszewski AJ,Desai A. Radiation therapy administration and survival in stageⅠ/Ⅱextranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue[J]. Int J Radiat Oncol Biol Phys,2014,88(3):642-649.
[28] Jackson AE,Mian M,Kalpadakis C,et al. Extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue of the salivary glands:a multicenter,international experience of 248 patients(IELSG 41)[J]. Oncologist,2015,20(10):1149-1153.
[29] Salles G,Barrett M,Foa R,et al. Rituximab in B-cell hematologic malignancies:a review of 20 years of clinical experience[J]. Adv Ther,2017,34(10):2232-2273.
[30]傅志英,朱军,宋玉琴,等. 525例弥漫大B细胞淋巴瘤预后影响因素分析[J].北京大学学报(医学版),2014,46(3):405-411.
[31]杨小芸,沈丽达,龙庭凤,等. 1 326例非霍奇金淋巴瘤临床病理特点分析[J].中华肿瘤防治杂志,2016,23(9):605-609.