Hepatic screlosed hemangioma which was misdiagnosed as metastasis of gastric cancer : report of a case

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1 270 CASE REPORT Hepti srelosed hemngiom whih ws misdignosed s metstsis of gstri ner : report of se Shinihiro Ymd, Mitsuo Shimd, Tohru Utsunomiy, Yuji Morine, Storu Imur, Tetsuy Ikemoto, Hiroki Mori, Jun Hnok, Shuihi Iwhshi, Yu Sitoh, nd Mihihito Asnom The Deprtment of Surgery, the University of Tokushim, Tokushim, Jpn Astrt : A srelosed hemngiom of the liver is rre mong hepti tumors. A 75 yers old mle ws referred to our hospitl for gstri ner nd hepti tumor. The histologil finding of gstri ner ws reveled to e well differentited denorinom. The liver tumor ws m in size nd loted in segment 8 of the liver. Computed tomogrphy (CT) showed it to e tumor with ring enhnement. Mgneti resonne imging (MRI) showed the tumor to hve low signl on T1-weighted nd slightly high signl on T2-weighted imges. Level of hemogloin ws 7.8 g/dl. It ws thought to e persistent leeding from gstri ner. With dignosis of liver metstsis from gstri ner, hemotherpy is reommended. However, to ontrol the leeding from gstri ner, we performed distl gstretomy nd wedge resetion of liver (S8). The histologil exmintion of the liver tumor reveled to e hepti slerosed hemngiom with hylinized tissue nd ollgen fiers. We report herein se of the rre tumor whih ws misdignosed s liver metstsis of gstri ner. J. Med. Invest. 59 : , August, 2012 Keywords : vernous hemngiom, liver, srelosed hemngiom, gstri ner, metstti tumor INTRODUCTION Cvernous hemngiom of the liver is the most ommon enign neoplsm (1). Cvernous hemngiom usully presents s solitry well-delineted, supsulr, nd dislosed nodules, nd these distintive strutures show hrteristi hemodynmi pttern on enhned omputed tomogrphy (CT). However, it ws lredy reported tht some ses ontin prts of hyline degenertion, seondry to thromus, nerosis, or itriztion, mimiking the hepti mlignnies suh s metstti liver tumor or holngiorinom (2). A hepti Reeived for pulition Mrh 8, 2012 ; epted April 11, Address orrespondene nd reprint requests to Mitsuo Shimd, MD, FACS, Deprtment of Surgery, the University of Tokushim, Kurmoto, Tokushim City, Tokushim, , Jpn nd Fx : slerosed hemngiom is the ultimte form hving ll these degenertive hnges (3). We report herein se of suh rre hepti tumor, slerosed hemngiom, whih ws misdignosed s liver metstsis of gstri ner. CASE REPORT A 75 yers old mle ws referred to our institute for gstri ner nd hepti spe oupied lesion (SOL). He hd visited n ffilited hospitl of our university for routine exmintion of prostti hyperplsi, nd ws pointed out nemi. For further exmintions, he took gstrosopy nd there ws type 3 tumor t posterior wll of gstri ngle (Fig 1). Biopsied speimens were dignosed s well differentited denorinom (tu1). Gstri fluorosopy showed tumor nd indurtion of lesser The Journl of Medil Investigtion Vol

2 The Journl of Medil Investigtion Vol. 59 August ompred to prenhym (iso-density) in the delyed phse (Fig 2). Mgneti resonne imging (MRI) showed tht the tumor hd low signl on T1-weighted nd high signl on T2-weighted imges. Heptoiliry phse of ethoxyenzyl (EOB)- enhned MRI showed no uptke in the orresponding re, s defet (Fig 3). Fluoro-deoxy-gluose positron emission tomogrphy (FDG-PET) showed Figure 1 : Gstri exmintions. () findings of gstrosopy. There is type 3 tumor t posterior wll in gstri ngle. The histologil exmintion reveled the tumor to e well differentited denorinom (tu1). () findings of Gstri fluorosopy. This shows tumor nd indurtion of the lesser urvture. urvture (Fig 1). Enhned CT showed smll mss in segment 8 of the liver, nd this hepti SOL ws suspeted to e metstsis of gstri ner. The initil lortory dt were s follows : 5300 /μl white lood ell ount, 7.8 g/dl hemogloin, 430,000 /μl pltelet ount, 19 IU/L sprtte minotrnsferse, 13 IU/L lnine minotrnsferse, 11 mg/dl lood ure nitrogen, 0.87 mg/dl retinin, 3.7 g/dl lumin. Tumor mrkers were s follows : 7 ng/ml lph-fetoprotein, 1.7 ng/ml rinoemryoni ntigen (CEA), nd 38 U/mL rohydrte ntigen (CA) He ws positive for heptitis B (HB) surfe ntiody (HBsA), HBeA nd HBA, nd negtive for HBsAg nd HBeAg. Plin CT showed low density mss, m in size, in segment 8 of the liver. Enhned CT showed tht the mss lesion ws grdully enhned minly in the mrginl order etween norml prenhym nd SOL, nd the mss ws not enhned Figure 2 : Adominl omputed tomogrphy. () Plin CT. () Contrst CT, erly phse. () Contrst CT delyed phse. Arrows indite the tumor. Figure 3 : Adominl mgneti resonne imging. () MRI, T1- weighted imge. () MRI, T2-weighted imge. () EOB enhned MRI, heptoyto phse. Arrows indite the tumor.

3 272 S. Ymd, et l. Hepti srelosed hemngiom tht FDG umulted only in gstri tumor (SUVmx 6.8). With these findings, we dignosed the liver mss s metstsis of gstri ner (Stge IV). In this se, hemotherpy is reommended ording to gstri ner tretment guideline. However, we performed distl gstretomy in order to ontrol the leeding from gstri ner. Opertive fingngs nd reseted speimens With fully informed onsents for possile hepti mlignnt tumor, we performed distl gstretomy. The edge of the liver ws shrp, nd liver surfe ws smooth. As intropertive ehogrphy reveled tht the liver mss of segment 8 ws ner the liver surfe, wedge resetion of segment 8 of the liver ws lso performed. The reseted speimen of the liver showed well demrted homogenous white solid mss, m in size, elsti hrd (Fig 4). Histrogil exmintions showed tht most re omprised hylinized tissue nd ollgen fiers with smll nd thin-wlled vsulr spe (Fig 5). Immunohistohemil stining reveled some vsulr omponents, s shown y positive CD34 in its peripherl region (Fig 5). Msson s trihrome stin highlighted the sleroti ondition of the mss Figure 4 : Mrosopi findings of the reseted tumor. The tumor ws well-demrted homogenous white solid nodule, m in size. () reseted speimen. () fter formlin preservtion. Figure 5 : Mirosopi findings of the reseted tumor. The tumor ws omposed of firous onnetive tissue nd vrious sizes of vernous hemngiom tissue, HE stining (), CD34 positive vsulr omponents (), Slerosis is highlighted with ollgen fier, Msson s trihrome ().

4 The Journl of Medil Investigtion Vol. 59 August (Fig 5). From these hrteristis, the tumor ws finlly dignosed s hepti slerosed hemngiom. Finl dignosis of gstri ner ws T3(ss), N2, ly2, v1, M0 (Stge III). DISCUSSION Cvernous hemngiom of the liver is the most frequent, the reported inidene eing s high s 20% in liver tumor (4). The high inidene of hemngiom inreses the proility of enountering typil mnifesttions, inluding nerosis, srring, nd lifition. A slerosed hemngiom is the ultimte form hving ll these degenertive hnges, nd suh vreties of pthologil hrteristis mke preisely rdiologil dignosis very diffiult (5). In our se, plin CT showed hepti srelosed hemnngiom s low density, nd enhned CT showed ring enhnement pttern, whih is hrteristi of denorinom. Although vernous hemngiom shows typil fetures : low density on plin CT nd delyed pooling enhnement, mny previous reports hve not shown this feture in slerosed hemngiom (5-12). MRI finding in our se ws fit to typil feture of vernous hemngiom, suh s low on T1 nd high on T2 weighted imges, ut this feture n e lso oserved in the ses of metstti liver tumors nd holngioellulr rinom. Others lso reported tht MRI findings of slerosed hemngiom were different from the typil vernous hemngiom (5-11). It ws reported tht nodulr res of redued signl intensity on T2- weighted imges orresponded to the histologil finding of firosis (13). A ompliting ftor is the ontriution to signl heterogeneity of hemorrhge, thromosis, hyliniztion, lifition, nd ysti vities (13). On the otherhnd, in our se, FDG umulted only in gstri tumor, not in hepti tumor in FDG-PET sn. However, the size of hepti tumor ws so smll tht this finding ould not eliminte the mlignny. With the ft tht there ws dvned gstri ner nd these rdiologil findings, metstti liver tumor from gstri ner ould not e exluded. It hs reported tht the findings suh s geogrphil pttern, psulr-retrtion, derese in size in follow-ups, nd vnishing of previous enhned lesion should rise the possiility of the presene of srelosed hemngiom, nd it n e dignosed with iopsy efore surgil intervention (5). However, we do not gree the universl pplition of pertneous needle iopsy to mke definite dignosis of hepti tumor suh s present se. Beouse perutneous iopsy hs een reported to hve potentil risk of dominl dissemintion y needle trt implnttion (14). As hepti resetion hs een sfe proedure in reent yers (15, 16), hepti resetion should e hosen s first strtegy for tumors with unknown pthologilly mlignnt potentil. Although slerosed hemngiom is rre disese mong hepti tumors, it needs to e tken into onsidertion mong differentil dignosis of hepti tumors. As it ws extremely diffiult to differentite the lesion from liver metstsis or holngioellrrinom, if the mlignny of the tumor nnot e denied, hepti resetion should e hosen for dignosti therpy. CONFLICT OF INTEREST STATEMENT The uthors delre no onflit of interest relted to this se report. REFERENCE 1. Aie H, Hondo H, Kuroiw T, Yoshimitsu K, Irie H, Tjim T, Shinozki K, Asym Y, Tguhi K, Msud K : Slerosed hemngiom of the liver. Adom Imging 26(5) : 496-9, Cheng HC, Tsi SH, Ching JH, Chng CY : Hylinized liver hemngiom mimiking mlignnt tumor t MR imging. AJR Am J Roentgenol 165(4) : , Mkhlouf HR, Ishk KG : Slerosed hemngiom nd slerosing vernous hemngiom of the liver : omprtive liniopthologi nd immunohistohemil study with emphsis on the role of mst ells in their histogenesis. Liver 22(1) : 70-8, Krhunen PJ : Benign hepti tumours nd tumour like onditions in men. J Clin Pthol 39 (2) : , Doyle DJ, Khlili K, Guindi M : Imging fetures of slerosed hemngiom. AJR Am J Roentgenol 189(1) : 67-72, Mthieu D, Rhmouni A, Vsile N, Jzerli N, Duvoux C, Trn JV, Zfrni ES : Slerosed liver hemngiom mimiking mlignnt tumor t MR imging : pthologi orreltion. J Mgn

5 274 S. Ymd, et l. Hepti srelosed hemngiom Reson Imging 4(3) : 506-8, Hrtke J, Horie A, Ngfuhi Y : Hylinized hemngiom of the liver. Am J Gstroenterol 87(2) : 234-6, Ymshit Y, Shimd M, Tguhi K, Gion T, Hsegw H, Utsunomiy T, Hmtsu T, Mtsumt T, Sugimhi K : Hepti slerosing hemngiom mimiking metstti liver tumor : report of se. Surg Tody 30(9) : , Shim KS, Suh JM, Yng YS, Kim JG, Kng SJ, Jeon JS, Kim BS : Slerosis of hepti vernous hemngiom : CT findings nd pthologi orreltion. J Koren Med Si 10(4) : 294-7, Tkysu K, Moriym N, Shim Y, Murmtsu Y, Ymd T, Mkuuhi M, Ymski S, Hirohshi S : Atypil rdiogrphi findings in hepti vernous hemngiom : orreltion with histologi fetures. AJR Am J Roentgenol 146(6) : , Vilgrin V, Boulos L, Vullierme MP, Denys A, Terris B, Menu Y : Imging of typil hemngioms of the liver with pthologi orreltion. Rdiogrphis 20(2) : , Mori H, Ikegmi T, Imur S, Shimd M, Morine Y, Knemur H, Arkw Y, Knmoto M, Hnok J, Sugimoto K, Tokung T : Slerosed hemngiom of the liver : Report of se nd review of the literture. Heptol Res 38(5) : , Ros PR, Luers PR, Olmsted WW, Morillo G : Hemngiom of the liver : heterogeneous pperne on T2-weighted imges. AJR Am J Roentgenol 149(6) : , Cturelli E, Ghittoni G, Roselli P, De Plo M, Anti M : Fine needle iopsy of fol liver lesions : the heptologist s point of view. Liver Trnspl 10(2 Suppl 1) : 26-9, Tsi MK, Lee PH, Tung BS, Yu SC, Lee CS, Wei TC : Experienes in surgil mngement of vernous hemngiom of the liver. Heptogstroenterology 42(6) : , Fn ST, Lo CM, Liu CL, Lm CM, Yuen WK, Yeung C, Wong J : Heptetomy for heptoellulr rinom : towrd zero hospitl deths. Ann Surg 229(3) : , 1999

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