罕见大隐静脉血管平滑肌肉瘤一例及文献荟萃分析
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摘要
大隐静脉平滑肌肉瘤(GSV-LMS)极为罕见,临床表现不典型,易发生误诊。本文报道了北京协和医院诊治的1例大隐静脉平滑肌肉瘤的病例资料,并与中国知网、万方数据库和MEDLINE数据库收录的41例大隐静脉平滑肌肉瘤资料共同进行荟萃分析。结果显示,本组病例男女比例为11∶10,发病中位年龄59.5岁;以大腿最好发,无特征性临床表现,42.9%患者在初诊时发生误诊。手术完整切除是目前最有效的治疗方式,辅助放化疗指征、效果及方案尚不明确。3年和5年总体生存率分别为86.1%和77.5%。31.0%的患者术后出现复发和转移。
Leiomyosarcoma(LMS)of the great saphenous vein(GSV)is a rare condition that is often misdiagnosed due to the lack of typical clinical manifestations.This article reported a GSV-LMS case diagnosed and treated in Peking Union Medical College Hospital.Meta-analysis of the current case and 41 cases derived from CNKI,WANFANG DATA,and MEDLINE database was also conducted,which revealed that the male to female ratio of GSV-LMS was 11∶10;the median age of disease onset was 59.5 years;the most commonly affected region was thigh.Due to lack of specific clinical features,42.9% of GSV-LMS patients were misdiagnosed at presentation.Radical resection is the most effective therapy,and the indications,effectiveness and protocols of adjuvant radiochemotherapy remain unclear.The 3-and 5-year overall survival rate was 86.1% and 77.5%,respectively.Recurrence and metastasis occurred in 31.0% of patients after surgery.
Leiomyosarcoma(LMS)of the great saphenous vein(GSV)is a rare condition that is often misdiagnosed due to the lack of typical clinical manifestations.This article reported a GSV-LMS case diagnosed and treated in Peking Union Medical College Hospital.Meta-analysis of the current case and 41 cases derived from CNKI,WANFANG DATA,and MEDLINE database was also conducted,which revealed that the male to female ratio of GSV-LMS was 11∶10;the median age of disease onset was 59.5 years;the most commonly affected region was thigh.Due to lack of specific clinical features,42.9% of GSV-LMS patients were misdiagnosed at presentation.Radical resection is the most effective therapy,and the indications,effectiveness and protocols of adjuvant radiochemotherapy remain unclear.The 3-and 5-year overall survival rate was 86.1% and 77.5%,respectively.Recurrence and metastasis occurred in 31.0% of patients after surgery.
引文
[1] Cangiano R,Orlandino G,Patitucci G,et al.Primary leiomyosarcoma of the great saphenous vein:case report and review of the literature[J].Ann Vasc Surg,2017,38:315.e1-e7.DOI:10.1016/j.avsg.2016.04.021.
[2] Gage MJ,Patel AV,Koenig KL,et al.Non-vena cava venous leiomyosarcomas:a review of the literature[J].Ann Surg Oncol,2012,19(11):3368- 3374.DOI:10.1245/s10434- 012- 2379- 2.
[3] Aufrecht E.Ein Myom der Vena saphena[J].Virchows Arch,1868,44(1):133- 135.DOI:10.1007/BF01959050.
[4] Fremed DI,Faries PL,Schanzer HR,et al.Primary leiomyosarcoma of saphenous vein presenting as deep venous thrombosis[J].Vascular,2014,22(6):450- 453.DOI:10.1177/1708538113516446.
[5] Fletcher CDM,Bridge JA,Hogendoorn P,et al.World Health Organization classification of tumours.Pathology and genetics of tumours of soft tissue and bone[M].4th ed.Lyon,France:IARC Press,2013.
[6] Wachtel H,Gupta M,Bartlett EK,et al.Outcomes after resection of leiomyosarcomas of the inferior vena cava:a pooled data analysis of 377 cases[J].Surg Oncol,2015,24(1):21- 27.DOI:10.1016/j.suronc.2014.10.007.
[7] Berlin O,Stener B,Kindblom LG,et al.Leiomyosarcomas of venous origin in the extremities.A correlated clinical,roentgenologic,and morphologic study with diagnostic and surgical implications[J].Cancer,1984,54(10):2147- 2159.
[8] Reix T,Sevestre H,Sevestri-Pietri MA,et al.Primary malignant tumors of the venous system in the lower extremities[J].Ann Vasc Surg,1998,12(6):589- 596.DOI:10.1007/s100169900205.
[9] Macarenco RS,Filippi RZ,D’Almeida CF,et al.Leiomyosarcoma of the great saphenous vein(vena saphena magna)with granular cell change:Report of a superficial neoplasm[J].J Cutan Pathol,2018,45(2):141- 145.DOI:10.1111/cup.13062.
[10] Zhang XS,Wang ZG.Primary leiomyosarcoma of the great saphenous vein:case report[J].Eur J Vasc Endovasc Surg,2006,32(2):222- 225.DOI:10.1016/j.ejvs.2005.12.018.
[11] Bibbo C,Schroeder M.Review of vascular leiomyosarcoma and report of a case localized to the greater saphenous vein of the ankle[J].J Foot Ankle Surg,2011,50(3):329- 335.DOI:10.1053/j.jfas.2011.01.002.
[12] 中国抗癌协会肉瘤专业委员会,中国临床肿瘤学会.软组织肉瘤诊治中国专家共识(2015版)[J].中华肿瘤杂志,2016,38(4):310- 320.DOI:10.3760/cma.j.issn.0253- 3766.2016.04.013.
[13] Teixeira FJ,Do CNS,Perina A,et al.Leiomyosarcoma of the inferior vena cava:Survival rate following radical resection[J].Oncol Lett,2017,14(4):3909- 3916.DOI:10.3892/ol.2017.6706.
[14] 李伟浩,张永保,李清乐,等.原发性下腔静脉平滑肌肉瘤12例回顾性分析[J].中华外科杂志,2015,53(9):690- 695.DOI:10.3760/cma.j.issn.0529- 5815.2015.09.013.
[15] Amin MB,Edge SB,Greene FL,et al.American Joint Committee on Cancer(AJCC)Cancer Staging Manual[M].8th ed.Switzterland:Springer International,2017.
[16] Humphrey M,Neff J,Lin F,et al.Leiomyosarcoma of the saphenous vein.A case report and review of the literature[J].J Bone Joint Surg Am,1987,69(2):282- 286.
[17] Marle AG,Bronkhorst MW,Brouwers MA.Leiomyosarcoma of the great saphenous vein:a case report and review of the literature[J].Sarcoma,2004,8(4):135- 139.DOI:10.1155/S1357714X04000234.
[18] Van Ree A.Phlebosarcoma racemosum[J].Ned Tijdschr Geneeskd,1919,63:759- 767.
[19] Deweese JA,Terry R,Schwartz SI.Leiomyoma of the greater saphenous vein with preoperative localization by phlebography[J].Ann Surg,1958,148(5):859- 861.DOI:10.1097/00000658- 195811000- 00027.
[20] Smout MS,Fisher JH.Leiomyosarcoma of the saphenous vein[J].Can Med Assoc J,1960,83(20):1066- 1067.
[21] Dorfman HD,Fishel ER.Report of a case and review of the literature[J].Am J Clin Pathol,1963,39(1):73- 78.
[22] Christiansen J.Malignant tumors originating from the venous system.Review and a case of leiomyosarcoma from the long saphenous vein[J].Ugeskr Laeger,1964,126:483- 485.
[23] Allison MF.Leiomyosarcoma of the femoral vein.Report of a case in a child[J].Clin Pediatr(Phila),1965,4:28- 31.
[24] Szasz IJ,Barr R,Scobie TK.Leiomyosarcoma arising from veins:two cases and a review of the literature on venous neoplasms[J].Can J Surg,1969,12(4):415- 419.
[25] Hughes S.A case of leiomyosarcoma of the long saphenous vein[J].Vasc Surg,1973,7(2):71- 73.
[26] Jernstrom P,Gowdy RA.Leiomyosarcoma of the long saphenous vein[J].Am J Clin Pathol,1975,63(1):25- 31.
[27] Gross E,Horton MA.Leiomyosarcoma of the saphenous vein[J].J Pathol,1975,116(1):37- 41.
[28] Stringer BD.Leiomyosarcoma of artery and vein[J].Am J Surg,1977,134(1):90- 94.
[29] Orsi G,Camera S,Zanlungo U.Primary peripheral venous tumors[J].Minerva Cardioangiol,1979,27(4):241- 244.
[30] Fischer MG,Gelb AM,Nussbaum M,et al.Primary smooth muscle tumors of venous origin[J].Ann Surg,1982,196(6):720- 724.
[31] Leu HJ,Makek M.Intramural venous leiomyosarcomas[J].Cancer,1986,57(7):1395- 1400.
[32] Song KY,Jang YW,Kim MK,et al.Leiomyosarcoma arising in the great saphenous vein-a case report[J].J Korean Med Sci,1991,6(4):372- 375.DOI:10.3346/jkms.1991.6.4.372.
[33] Welk E,Bindewald H,Berndt R.Vascular leiomyosarcoma-a surgical rarity[J].Chirurg,1991,62(3):223- 225.
[34] Dzsinich C,Gloviczki P,van Heerden JA,et al.Primary venous leiomyosarcoma:a rare but lethal disease[J].J Vasc Surg,1992,15(4):595- 603.DOI:10.1016/0741- 5214(92)90003-q.
[35] Saglik Y,Icli F,Uluoglu O,et al.Leiomyosarcoma of the great saphenous vein[J].Int Orthop,1992,16(2):185- 187.
[36] Stallard D,Sundaram M,Johnson FE,et al.Case report 747:Leiomyosarcoma of great saphenous vein[J].Skeletal Radiol,1992,21(6):399- 401.
[37] Stambuk J,Oddo D,Perez R,et al.Leiomyosarcoma of the saphenous vein[J].Rev Med Chil,1993,121(6):673- 676.
[38] Byard RW,Bourne AJ,Phillips GE,et al.Leiomyosarcoma of the saphenous vein in a child with 12-year follow-up[J].J Pediatr Surg,1993,28(2):271- 274.DOI:10.1016/s0022- 3468(05)80294- 5.
[39] Le Minh T,Cazaban D,Michaud J,et al.Great saphenous vein leiomyosarcoma:a rare malignant tumor of the extremity-two case reports[J].Ann Vasc Surg,2004,18(2):234- 236.DOI:10.1007/s10016- 003- 0090- 2.
[40] El Khoury M,Mesurolle B,Trassard M,et al.Leiomyosarcoma of the great saphenous vein[J].Australas Radiol,2006,50(5):500- 503.DOI:10.1111/j.1440- 1673.2006.01636.x.
[41] Mammano E,Zanon A,Picchi G,et al.Primary great saphenous vein leiomyosarcoma:report of a case[J].Surg Today,2008,38(2):161- 162.DOI:10.1007/s00595- 007- 3588- 3.
[42] Yanada M,Shimada J,Kohnosu H.A case of primary great saphenous vein leiomyosarcoma showing high uptake on FDG-PET[J].Nihon Rinsho Geka Gakkai Zasshi,2010,71(6):1663- 1666.DOI:10.3919/jjsa.71.1663.
[43] Amato B,Compagna R,Gasbarro V,et al.Great saphenous vein and leiomyosarcoma[J].EJVES Extra,2013,26(1):e15-e16.DOI:10.1016/j.ejvsextra.2013.03.001.
[44] 徐翠,刘灿,杨智,等.大隐静脉内平滑肌肉瘤超声表现1例[J].中华超声影像学杂志,2013,22(5):382.DOI:10.3760/cma.j.issn.1004- 4477.2013.05.006.
[45] Lin WW,Erkan S,Smith RC,et al.Leiomyosarcoma of great saphenous vein localised to the calf[J].BMJ Case Rep,2016,2016:r2016215829.DOI:10.1136/bcr- 2016- 215829.
[2] Gage MJ,Patel AV,Koenig KL,et al.Non-vena cava venous leiomyosarcomas:a review of the literature[J].Ann Surg Oncol,2012,19(11):3368- 3374.DOI:10.1245/s10434- 012- 2379- 2.
[3] Aufrecht E.Ein Myom der Vena saphena[J].Virchows Arch,1868,44(1):133- 135.DOI:10.1007/BF01959050.
[4] Fremed DI,Faries PL,Schanzer HR,et al.Primary leiomyosarcoma of saphenous vein presenting as deep venous thrombosis[J].Vascular,2014,22(6):450- 453.DOI:10.1177/1708538113516446.
[5] Fletcher CDM,Bridge JA,Hogendoorn P,et al.World Health Organization classification of tumours.Pathology and genetics of tumours of soft tissue and bone[M].4th ed.Lyon,France:IARC Press,2013.
[6] Wachtel H,Gupta M,Bartlett EK,et al.Outcomes after resection of leiomyosarcomas of the inferior vena cava:a pooled data analysis of 377 cases[J].Surg Oncol,2015,24(1):21- 27.DOI:10.1016/j.suronc.2014.10.007.
[7] Berlin O,Stener B,Kindblom LG,et al.Leiomyosarcomas of venous origin in the extremities.A correlated clinical,roentgenologic,and morphologic study with diagnostic and surgical implications[J].Cancer,1984,54(10):2147- 2159.
[8] Reix T,Sevestre H,Sevestri-Pietri MA,et al.Primary malignant tumors of the venous system in the lower extremities[J].Ann Vasc Surg,1998,12(6):589- 596.DOI:10.1007/s100169900205.
[9] Macarenco RS,Filippi RZ,D’Almeida CF,et al.Leiomyosarcoma of the great saphenous vein(vena saphena magna)with granular cell change:Report of a superficial neoplasm[J].J Cutan Pathol,2018,45(2):141- 145.DOI:10.1111/cup.13062.
[10] Zhang XS,Wang ZG.Primary leiomyosarcoma of the great saphenous vein:case report[J].Eur J Vasc Endovasc Surg,2006,32(2):222- 225.DOI:10.1016/j.ejvs.2005.12.018.
[11] Bibbo C,Schroeder M.Review of vascular leiomyosarcoma and report of a case localized to the greater saphenous vein of the ankle[J].J Foot Ankle Surg,2011,50(3):329- 335.DOI:10.1053/j.jfas.2011.01.002.
[12] 中国抗癌协会肉瘤专业委员会,中国临床肿瘤学会.软组织肉瘤诊治中国专家共识(2015版)[J].中华肿瘤杂志,2016,38(4):310- 320.DOI:10.3760/cma.j.issn.0253- 3766.2016.04.013.
[13] Teixeira FJ,Do CNS,Perina A,et al.Leiomyosarcoma of the inferior vena cava:Survival rate following radical resection[J].Oncol Lett,2017,14(4):3909- 3916.DOI:10.3892/ol.2017.6706.
[14] 李伟浩,张永保,李清乐,等.原发性下腔静脉平滑肌肉瘤12例回顾性分析[J].中华外科杂志,2015,53(9):690- 695.DOI:10.3760/cma.j.issn.0529- 5815.2015.09.013.
[15] Amin MB,Edge SB,Greene FL,et al.American Joint Committee on Cancer(AJCC)Cancer Staging Manual[M].8th ed.Switzterland:Springer International,2017.
[16] Humphrey M,Neff J,Lin F,et al.Leiomyosarcoma of the saphenous vein.A case report and review of the literature[J].J Bone Joint Surg Am,1987,69(2):282- 286.
[17] Marle AG,Bronkhorst MW,Brouwers MA.Leiomyosarcoma of the great saphenous vein:a case report and review of the literature[J].Sarcoma,2004,8(4):135- 139.DOI:10.1155/S1357714X04000234.
[18] Van Ree A.Phlebosarcoma racemosum[J].Ned Tijdschr Geneeskd,1919,63:759- 767.
[19] Deweese JA,Terry R,Schwartz SI.Leiomyoma of the greater saphenous vein with preoperative localization by phlebography[J].Ann Surg,1958,148(5):859- 861.DOI:10.1097/00000658- 195811000- 00027.
[20] Smout MS,Fisher JH.Leiomyosarcoma of the saphenous vein[J].Can Med Assoc J,1960,83(20):1066- 1067.
[21] Dorfman HD,Fishel ER.Report of a case and review of the literature[J].Am J Clin Pathol,1963,39(1):73- 78.
[22] Christiansen J.Malignant tumors originating from the venous system.Review and a case of leiomyosarcoma from the long saphenous vein[J].Ugeskr Laeger,1964,126:483- 485.
[23] Allison MF.Leiomyosarcoma of the femoral vein.Report of a case in a child[J].Clin Pediatr(Phila),1965,4:28- 31.
[24] Szasz IJ,Barr R,Scobie TK.Leiomyosarcoma arising from veins:two cases and a review of the literature on venous neoplasms[J].Can J Surg,1969,12(4):415- 419.
[25] Hughes S.A case of leiomyosarcoma of the long saphenous vein[J].Vasc Surg,1973,7(2):71- 73.
[26] Jernstrom P,Gowdy RA.Leiomyosarcoma of the long saphenous vein[J].Am J Clin Pathol,1975,63(1):25- 31.
[27] Gross E,Horton MA.Leiomyosarcoma of the saphenous vein[J].J Pathol,1975,116(1):37- 41.
[28] Stringer BD.Leiomyosarcoma of artery and vein[J].Am J Surg,1977,134(1):90- 94.
[29] Orsi G,Camera S,Zanlungo U.Primary peripheral venous tumors[J].Minerva Cardioangiol,1979,27(4):241- 244.
[30] Fischer MG,Gelb AM,Nussbaum M,et al.Primary smooth muscle tumors of venous origin[J].Ann Surg,1982,196(6):720- 724.
[31] Leu HJ,Makek M.Intramural venous leiomyosarcomas[J].Cancer,1986,57(7):1395- 1400.
[32] Song KY,Jang YW,Kim MK,et al.Leiomyosarcoma arising in the great saphenous vein-a case report[J].J Korean Med Sci,1991,6(4):372- 375.DOI:10.3346/jkms.1991.6.4.372.
[33] Welk E,Bindewald H,Berndt R.Vascular leiomyosarcoma-a surgical rarity[J].Chirurg,1991,62(3):223- 225.
[34] Dzsinich C,Gloviczki P,van Heerden JA,et al.Primary venous leiomyosarcoma:a rare but lethal disease[J].J Vasc Surg,1992,15(4):595- 603.DOI:10.1016/0741- 5214(92)90003-q.
[35] Saglik Y,Icli F,Uluoglu O,et al.Leiomyosarcoma of the great saphenous vein[J].Int Orthop,1992,16(2):185- 187.
[36] Stallard D,Sundaram M,Johnson FE,et al.Case report 747:Leiomyosarcoma of great saphenous vein[J].Skeletal Radiol,1992,21(6):399- 401.
[37] Stambuk J,Oddo D,Perez R,et al.Leiomyosarcoma of the saphenous vein[J].Rev Med Chil,1993,121(6):673- 676.
[38] Byard RW,Bourne AJ,Phillips GE,et al.Leiomyosarcoma of the saphenous vein in a child with 12-year follow-up[J].J Pediatr Surg,1993,28(2):271- 274.DOI:10.1016/s0022- 3468(05)80294- 5.
[39] Le Minh T,Cazaban D,Michaud J,et al.Great saphenous vein leiomyosarcoma:a rare malignant tumor of the extremity-two case reports[J].Ann Vasc Surg,2004,18(2):234- 236.DOI:10.1007/s10016- 003- 0090- 2.
[40] El Khoury M,Mesurolle B,Trassard M,et al.Leiomyosarcoma of the great saphenous vein[J].Australas Radiol,2006,50(5):500- 503.DOI:10.1111/j.1440- 1673.2006.01636.x.
[41] Mammano E,Zanon A,Picchi G,et al.Primary great saphenous vein leiomyosarcoma:report of a case[J].Surg Today,2008,38(2):161- 162.DOI:10.1007/s00595- 007- 3588- 3.
[42] Yanada M,Shimada J,Kohnosu H.A case of primary great saphenous vein leiomyosarcoma showing high uptake on FDG-PET[J].Nihon Rinsho Geka Gakkai Zasshi,2010,71(6):1663- 1666.DOI:10.3919/jjsa.71.1663.
[43] Amato B,Compagna R,Gasbarro V,et al.Great saphenous vein and leiomyosarcoma[J].EJVES Extra,2013,26(1):e15-e16.DOI:10.1016/j.ejvsextra.2013.03.001.
[44] 徐翠,刘灿,杨智,等.大隐静脉内平滑肌肉瘤超声表现1例[J].中华超声影像学杂志,2013,22(5):382.DOI:10.3760/cma.j.issn.1004- 4477.2013.05.006.
[45] Lin WW,Erkan S,Smith RC,et al.Leiomyosarcoma of great saphenous vein localised to the calf[J].BMJ Case Rep,2016,2016:r2016215829.DOI:10.1136/bcr- 2016- 215829.