Case Report Surgical Resection of a Leiomyosarcoma of the Inferior Vena Cava Mimicking Hepatic Tumor

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1 Cse Reports in Medicine Volume 2013, Article ID , 5 pges Cse Report Surgicl Resection of Leiomyosrcom of the Inferior Ven Cv Mimicking Heptic Tumor Junji Ued, 1 Hiroshi Yoshid, 1 Ysuhiro Mmd, 1 Nouhiko Tnii, 1 Msto Yoshiok, 1 Youichi Kwno, 1 Yoshiki Mizuguchi, 2 Tetsuy Shimizu, 1 Hideyuki Tkt, 1 nd Eiji Uchid 1 1 Deprtment of Gstrointestinl Heptoiliry-Pncretic Surgery, Nippon Medicl School, Tokyo , Jpn 2 Deprtment of Surgery for Orgn Function nd Biologicl Regultion, Grdute School of Medicine, Nippon Medicl School, Tokyo, Jpn Correspondence should e ddressed to Junji Ued; junji0821@nms.c.jp Received 8 Octoer 2012; Accepted 20 Jnury 2013 Acdemic Editor: John A. Elefterides Copyright 2013 Junji Ued et l. This is n open ccess rticle distriuted under the Cretive Commons Attriution License, which permits unrestricted use, distriution, nd reproduction in ny medium, provided the originl work is properly cited. Introduction. Leiomyosrcoms of vsculr origin re prticulrly rre tumors occurring minly in the inferior ven cv (IVC). They re mlignnt, slow-growing tumors with poor prognosis. This pper reports on rre cse of surgicl resection of n IVC leiomyosrcom mimicking heptic tumor. Cse Presenttion. A 65-yer-old Jpnese mle ws dmitted for evlution of n dominl tumor. Enhnced computed tomogrphy of the domen reveled slightly enhnced heterogeneous tumor, 18 mm in dimeter, etween the Spiegel loe of the liver nd the IVC in erly-phse imges, with no enhncement or wshout in lte-phse imges. We dignosed this tumor s either heptic tumor in the Spiegel loe or retroperitonel tumor such s leiomyosrcom or liposrcom nd performed lprotomy. On the sis of surgicl findings, we extirpted the tumor y performing wedge resection of the wll of the IVC nd suturing the primry IVC wll. Pthologicl findings led to further dignosis of the tumor s leiomyosrcom originting in the IVC. Thirty-seven months fter the opertion, multiple liver nd lung metstses were detected, nd the ptient died from multiple orgnic filures. Conclusion. We experienced rre cse of leiomyosrcom of IVC mimicking heptic tumor. 1. Introduction Leiomyosrcoms re mlignnt tumors of smooth muscle cells tht cn originte in ny loction ut occur most often in the uterus, retroperitoneum, or intrdominl region [1]. Leiomyosrcoms of vsculr origin re prticulrly rre tumors occurring minly in the inferior ven cv (IVC) [2, 3]. The Interntionl Registry of Inferior Ven Cv Leiomyosrcoms reveled tht most tumors occur in the lower (44.2%) or middle (50.8%) region of the IVC, while only smll numer of tumors occur in the upper third or suprheptic region (4.2%) [4]. Leiomyosrcom of IVC ws first descried y Dzsinich et l. in 1992 [5]. There re no definitive symptoms, ut typicl symptoms include dyspne nd generl feeling of eing unwell ccompnied y weight loss nd dominl nd/or ck pin [6]. A correct dignosis is difficult, nd, in up to one-third of cses, it is not mde until fter postmortem exmintion [3]. Dignosis is mde using modern imging techniques such s ultrsound, echocrdiogrphy, computed tomogrphy (CT), nd mgnetic resonnce imging (MRI) [2]. In spite of the dvncement of modern imging techniques, it is difficult to dignose leiomyosrcom preopertively, nd in some cses it is lso difficult to distinguish it from heptic tumor [4]. This pper reports on rre cse of surgicl resection of n IVC leiomyosrcom mimicking heptic tumor. 2. Cse Presenttion A 65-yer-old Jpnese mle ws dmitted for evlution of n dominl tumor. The tumor ws detected y chnce during chest CT performed for lung exmintion. The ptient hd no pst history of ny dominl surgery, nd

2 2 Cse Reports in Medicine Figure 1: Enhnced computed tomogrphy (CT) of the domen reveled slightly enhnced heterogeneous tumor, 18 mm in dimeter, etween the Spiegel loe of the liver nd the IVC in erly-phse imges ( rrow), with no enhncement nd wshout in lte-phse imges ( rrow). Figure 2: Mgnetic resonnce imging (MRI) of the domen. Mgnetic resonnce imging (MRI) of this tumor reveled contrsting low intensity on the T1-weighted imge ( rrow hed) nd high intensity on the T2-weighted imge ( rrow hed). his vitl signs were stle. There is nothing prticulrly significnt in the findings of the lortory exmintion. Tumor mrkers re negtive. Enhnced computed tomogrphy of the domen reveled slightly enhnced heterogeneous tumor, 18 mm in dimeter, etween the Spiegel loe of the liver nd the IVC in erly-phse imges (Figure 1), with no enhncement or wshout in lte-phse imges (Figure 1). Mgnetic resonnce imging of this tumor reveled contrsting low intensity on the T1-weighted imge (Figure 2) nd high intensity on the T2-weighted imge (Figure 2). Upper-gstrointestinl endoscopy nd colonoscopy reveled no evidence of mlignnt tumor in the gstrointestinl trct. We dignosed this tumor s either heptic tumor in the Spiegel loe or retroperitonel tumor such s leiomyosrcom or liposrcom. A lprotomy ws performed, nd the surfce of the tumor ws found to e smooth nd slightly dhesive. To expose the ffected IVC lesion, we need to moilize some prts of the right loe of the liver. This tumor ws not locted in the liver ut originted in the IVC. We extirpted the tumor y performing wedged resection of the wll of the IVC nd sutured primry IVC wll. We controlled the leedingfromtheivcusingthehemostticforceps.the resected specimen ws solid with smooth surfce (Figure 3). Microscopic exmintion reveled tht the tumor consisted of uniform nd spindle cells nd hd fsciculr growth pttern. This pthologicl feture ws comptile with mesenchyml tumor (Figure 4). Immunohistochemicl stining reveled tht α-sma nd HHF35 were expressed in this tumor (Figures 4 nd 4(c)), nd c-kit nd CD34 were negtive. The MIB-1 index ws out 60% (Figure 4(d)). The tumor ws dignosed s leiomyosrcom originting in the IVC. The ptient s recovery ws uneventful, nd he ws dischrged on postopertive dy 10. After 12 months, the tumorwsdetectedinthespiegeloftheliveronenhncedct of the domen (Figure 5). We dignosed it s recurrence of the leiomyosrcom. We suggested to the ptient nd his fmily tht he should undergo dditionl tretments such s chemotherpy, rdition, or surgicl resection, ut they declined this. Thirty-seven months fter the opertion, multiple liver nd lung metstses were detected on CT (Figures 6 nd 6), nd the ptient died of multiple orgnic filures. 3. Discussion Leiomyosrcoms originting in the IVC re rre, mlignnt, slow-growing tumors with poor prognosis [6]. Tumor metstses of this slow-growing mlignnt tumor to other

3 Cse Reports in Medicine 3 Figure 3: The resected specimen ws solid with smooth surfce. c d (c) (d) Figure 4: Histopthologicl findings. Microscopic exmintion reveled tht the tumor consisted of uniform nd spindle cells nd hd fsciculr growth pttern (Hemtoxylin & Eosin: 600). Immunohistochemicl stining reveled tht α-sma ( 600) nd HHF35 ( 600) (c) were expressed in this tumor; MIB-1 index ws out 60% (Ki67: 600) (d). orgns re reltively infrequent, ut cn e detected in the liver, lungs, lymph nodes, or ones [7, 8]. In this cse, there were no symptoms, nd the tumor ws detected y chnce in chest CT. Present dy imging techniques such s dominl ultrsonogrphy, echocrdiogrphy, CT, nd MRI re commonly employed to mke rpid nd precise dignosis of suspected leiomyosrcom [6]. In this cse we could not preopertively dignose the tumor s leiomyosrcom of theivcndlsocouldnotruleoutpotentillivertumor. A finl conclusive dignosis is of course mde y mens of histopthologicl nd immunohistochemicl methods. The pthognomonic findings of leiomyosrcom re spindleshped tumor cells with positive mrkers for smooth muscle cells, vimentin, muscle ctin, lph-smooth muscle ctin, nd desmin [9]. The recommended therpy for treting leiomyosrcom is ggressive surgicl removl of the tumor y mens of modern vsculr surgery, in comintion with chemotherpy

4 4 Cse Reports in Medicine Figure 5: Enhnced computed tomogrphy (CT) of the domen. The tumor ws detected in the Spiegel of the liver on enhnced CT of the domen (rrow). Figure 6: Computed tomogrphy (CT) of the domen nd chest. Multiple liver metstses nd lung metstses ( rrows) were detected on CT. nd/or rdiotherpy [2, 8, 10]. In surgery, complete resection of the tumor ws possile, nd the IVC ws repired primrily vi surgicl mens without too high risk of postopertive edem. For leiomyosrcoms, there is periopertive mortlity of 4%, nd 42% of the ptients died of the disese itself [11]. To reduce the tumor size nd increse the resection rte, preopertive neodjuvnt therpy cn e implemented. If, however, complete tumor resection is not possile, tumor reduction followed y rdition therpy provides good pllitive tretment option [12]. Postopertive therpy nd the mngement of recurrence redifficultecuseofthesenceofevidencefortheir effectiveness in ptients. Rdition hs een used in oth the neodjuvnt nd djuvnt settings, nd some uthors elieve tht it my help with the locl control of disese [13]. For loclized, resectle, soft-tissue srcoms, djuvnt chemotherpy with doxoruicin or comintion of doxoruicin nd ifosfmide hs een shown to prolong the time efore recurrence nd the overll rte of survivl [14]. Thistherpymythuslsoeeffectiveinthetretmentof IVC leiomyosrcom. A cse hs een previously reported in which n IVC leiomyosrcom with liver metstsis ws positive for steroid receptors, including the estrogen receptor (ER) nd progesterone receptor (PR) [4]. The ptient received comined multimodl therpy, including resection of the tumor, intrheptic rteril chemotherpy, nd hormonl mnipultion. Thus hormonl mnipultion using medroxyprogesterone nd/or tmoxifen should e tried in ER/PRpositive cses. Although there is no estlished tretment for ptients with IVC leiomyosrcom, we hope tht thorough investigtion of this condition will help estlish stndrd tretment yielding stisfctory results [4]. In series of cse studies of 14 ptients with leiomyosrcom of the IVC who underwent generous resection followed y rdition, it ws estlished tht rdiotherpy reduced locl recurrence nd incresed the medin survivl time. In ddition, comintion of chemotherpy nd rdiotherpy hs een reported s eing superior to rdiotherpy lone with respect to n increse in the survivl rte [13]. The longterm outcome of surgery for leiomyosrcom of the IVC hs een disppointing. Some reports with sufficient cse numers nd followup reveled tht 5-yer survivl rte

5 Cse Reports in Medicine 5 ws chieved 33% 53% fter rdicl resection followed y curtive pproch using djuvnt therpy [2, 8, 13]. We should dignose this tumor in erly stge, nd ggressive surgicl mngement using modern vsculr surgicl nd oncology techniques. 4. Conclusion We encountered rre cse of leiomyosrcom of the IVC mimicking liver tumor. We must lwys consider leiomyosrcom when treting tumor etween the liver nd the IVC nd e prepred to perform complete surgicl resection in the event of leiomyosrcom nd follow up crefully. We must lso consider djuvnt therpy for recurrent leiomyosrcom. Authors Contriution J. Ued, H. Yoshid, nd E. Uchid contriuted eqully to this work; Y. Mmd, N. Tnii, M. Yoshiok, Y. Kwno, T. Shimizu, nd H. Tkt performed the opertion. All uthors red nd pproved the finl pper. Acknowledgment The Deprtment of Surgery for Orgn nd Biologicl Regultion, Nippon Medicl School, performed the histologicl exmintion of the leiomyosrcom nd plyed crucil role in producing this pper. inferior ven cv leiomyosrcom, the Americn College of Surgeons,vol.197,no.4,pp ,2003. [9] T.Nikido,Y.Endo,S.Nimur,H.Ishikur,ndS.Ushigome, Dumell-shped leiomyosrcom of the inferior ven cv with foci of rhdoid chnges nd osteoclst-type gint cells, Pthology Interntionl, vol. 54, no. 4, pp , [10] E. Crem, M. G. Znier Gomes, I. D. O. Monteiro, T. S. de Lim, nd A. A. Silv, Leiomyosrcom of the inferior ven cv: cse report, Angiology,vol.59,no.2,pp ,2008. [11] N. J. Hillird, M. J. Heslin, nd C. Y. Cstro, Leiomyosrcom of the inferior ven cv: three cse reports nd review of the literture, Annls of Dignostic Pthology,vol.9,no.5,pp , [12]D.Hemnt,R.Krntikumr,J.Amit,A.Chwl,ndN. Rnjeet, Primry leiomyosrcom of inferior ven cv, rre entity: imging fetures, Austrlsin Rdiology,vol.45,no.4, pp ,2001. [13] O. J. Hines, S. Nelson, W. J. Quinones-Bldrich, nd F. R. Eiler, Leiomyosrcom of the inferior ven cv: prognosis nd comprison with leiomyosrcom of other ntomic sites, Cncer,vol.85,no.5,pp ,1999. [14] N. Perviz, N. Colterjohn, F. Frrokhyr, R. Tozer, A. Figueredo, nd M. Ghert, A systemtic met-nlysis of rndomized controlled trils of djuvnt chemotherpy for loclized resectle soft-tissue srcom, Cncer,vol.113,no.3,pp ,2008. References [1] G. Ris, S. Rissouni, H. Mouzount et l., Primry pleurl leiomyosrcom with rpid progression nd ftl outcome: cse report, Medicl Cse Reports,vol.6,no.1,rticle 101, [2] E. Kieffer, M. Aloui, J. C. Piette, P. Ccou, nd L. Chiche, Leiomyosrcom of the inferior ven cv: experience in 22 cses, Annls of Surgery,vol.244,no.2,pp ,2006. [3] W. B. Lskin, J. C. Fnurg-Smith, A. P. Burke, E. Krszewsk, J. F. Fetsch, nd M. Miettinen, Leiomyosrcom of the inferior ven cv: clinicopthologic study of 40 cses, The Americn Surgicl Pthology,vol.34,no.6,pp ,2010. [4] M. Hshimoto, T. Koyshi, H. Tshiro et l., A huge metsttic liver tumor from leiomyosrcom of the inferior vencv:reportofcse, Surgery Tody, vol.42,no.5,pp , [5] C. Dzsinich, P. Gloviczki, J. A. vn Heerden et l., Primry venous leiomyosrcom: rre ut lethl disese, Vsculr Surgery, vol. 15, no. 4, pp , [6] U. Lotze, J. Reponov, G. Muth et l., Leiomyosrcom of the inferior ven cv extending into the right trium: rre differentil dignosis of right tril tumor with ftl outcome, Herz, vol. 37, no. 5, pp , [7] A. S. Griffin nd J. M. Sterchi, Primry leiomyosrcom of the inferior ven cv: cse report nd review of the literture, Surgicl Oncology,vol.34,no.1,pp.53 60,1987. [8] S.T.Holleneck,S.R.Gromyer,K.C.Kent,ndM.F.Brennn, Surgicl tretment nd outcomes of ptients with primry

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