Multiple malignant epithelioid mesotheliomas of the liver and greater omentum: a case report and review of the literature

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1 Minmi et l. Surgil Cse Reports (2017) 3:66 DOI /s y CASE REPORT Multiple mlignnt epithelioid mesothelioms of the liver nd greter omentum: se report nd review of the literture Open Aess Koji Minmi 1, Hiroshi Okumur 1*, Kiyokzu Hiwtshi 1, Sumik Mtsukit 2, Tetsuro Setoym 1, Kot Minmimgri 1, Yusuke Tsurut 1, Ihiro Knetsuki 3, Yoshito Ogur 4, Shigeho Menohr 1 nd Shoji Ntsugoe 4 Astrt Bkground: Mlignnt mesotheliom ommonly rises from the pleur, ut n lso rise from the peritoneum, perirdium, nd tuni vginlis testis. However, mlignnt mesotheliom of the liver is extremely rre nd oexistene with mlignnt mesotheliom of the greter omentum hs not een desried in the literture. In this se report, we present se of multiple mlignnt mesotheliom of the liver nd greter omentum. Cse presenttion: A 36-yer-old womn ws dmitted to our hospitl for the evlution of n elsti hrd mss in the right upper domen. Adominl ontrst omputed tomogrphy showed ysti mss mesuring m in the right liver loe with enhned murl nodule. Anorml umultion ws identified in the liver nd lower dominl re on 18 F-fluorodeoxygluose positron emission tomogrphy. The ptient underwent heptetomy of the posterior segment nd prtil resetion of the omentum. The finl pthologil dignosis ws low-grde multiple mlignnt epithelioid mesotheliom sed on hrteristi immunohistohemil findings. As of 6 months postopertively, the ptient hs shown no disese reurrene. Conlusions: We present the first se of 36-yer-old womn with multiple mlignnt mesotheliom of the liver nd greter omentum. Keywords: Multiple mlignnt mesotheliom, Liver, Greter omentum, Epithelioid type Bkground Mlignnt mesotheliom ommonly rises from the mesothelil surfes of the pleurl vities nd is usully ssoited with inhltion of sestos fiers [1]. In ontrst, peritonel mlignnt mesotheliom is rre disese, nd mlignnt mesotheliom rising in the liver is extremely rre [2 12]. Moreover, mlignnt mesotheliom of the greter omentum is lso extremely rre [13, 14]. We present herein the se of 36-yer-old womn with multiple mlignnt mesotheliom of the liver nd greter omentum. * Correspondene: hokumur@m.kufm.kgoshim-u..jp 1 Deprtment of Surgery, Kgoshim Kouseiren Hospitl, Tenpozn 22-25, Kgoshim , Jpn Full list of uthor informtion is ville t the end of the rtile Cse presenttion A 36-yer-old womn with 2-month history of dominl nd k pin ws referred nd dmitted to our hospitl for tretment. The ptient hd no history of disese. On physil exmintion, hrd, elsti, poorly flexile mss ws plple in the right upper domen. No signs of ostrutive jundie or dominl tenderness were identified. Lortory nlysis reveled nemi (hemogloin = 10.6 g/dl), thromoytosis ( / μl), nd elevtion of C-retive protein (2.71 mg/dl). The remining lortory exmintions were within norml rnges, inluding tumor mrkers α-fetoprotein (AFP), rinoemryoni ntigen (CEA), nd ner ntigen (CA)19-9. Adominl ultrsonogrphy reveled n extensive spe-oupying lesion in the right loe of the liver, 15 m in dimeter, showing heterogeneous The Author(s) Open Aess This rtile is distriuted under the terms of the Cretive Commons Attriution 4.0 Interntionl Liense ( whih permits unrestrited use, distriution, nd reprodution in ny medium, provided you give pproprite redit to the originl uthor(s) nd the soure, provide link to the Cretive Commons liense, nd indite if hnges were mde.

2 Minmi et l. Surgil Cse Reports (2017) 3:66 internl omponent inluding hemorrhge nd hypervsulr murl nodule (Fig. 1, ). Contrst-enhned omputed tomogrphy (CT) of the domen showed ysti mss mesuring m in the right loe of the liver with enhned murl nodule (Fig. 1). Mgneti resonne imging (MRI) of the domen showed hyperintense omponents on T2-weighted imging, omptile with the hemorrhgi re (Fig. 1d). Anorml umultion ws seen on 18F-fluorodeoxygluose positron emission tomogrphy (FDG-PET) of the liver nd lower domen (Fig. 2). We then plnned exision of the tumor nd performed heptetomy of the posterior segment nd prtil resetion of the omentum, whih hd een deteted on FDG-PET. Gross exmintion of the hepti tumor showed mssive ysti tumor mesuring m ontining hemorrhgi fluid, nd muinous, hemorrhgi rownish or yellowish multinodulr tumors were oserved in the extr ysti wll re (Fig. 3, ). The omentl tumors were two slightly rownish, nodulr tumors mesuring nd m (Fig. 3). Mirosopilly, oth hepti nd omentl tumors omprised tuulr, ysti, or spindle strutures of epithelioid ells with ler or eosinophili ytoplsm (Fig. 4, ). Immunohistohemilly, tumor ells were positive for AE1/AE3, EMA, CK19, CK7, CD10, nd lretinin (Fig. 4), prtly or wekly positive for CK5/6, D2-40, vimentin, nd WT-1, nd negtive for Pge 2 of 5 HepPer1, hromogrnin A, synptophysin, CEA, inhiin α, MUC1, meln A, HMB45, CA19-9, ER, PgR, CD34, l-2, nd β-tenin (Fig. 4d, e). Ki-67 index ws 5 6% (Fig. 4f ). The finl pthologil dignosis ws multiple low-grde mlignnt epithelioid mesotheliom. As of the time of writing, 6 months postopertively, the ptient hs shown no disese reurrene. Disussion Mlignnt mesotheliom ommonly rises from the pleur, ut n lso rise from the peritoneum, perirdium, nd tuni vginlis testis [2]. However, mlignnt mesotheliom of the liver is extremely rre nd oexistene with mlignnt mesotheliom of the greter omentum hs not een ddressed in the literture. Mesotheliom of the liver my rise from Glisson s psule, the hepti fliform ligment or firous onnetive tissue, nd then expnd to the liver prenhym [3]. To serh the litertures, we used key words of intrhepti, mlignnt, nd mesotheliom nd found 12 se reports whih hve desried primry intrhepti mlignnt mesotheliom [2 12] (Tle 1). In detil, these ses involved six men nd six women, with men ge of 58.4 yers (rnge, yers). Our se represents the youngest femle se mong these reported ses. Although onventionl mesotheliom is frequently ssoited with sestos exposure, only 1 se hd ler d Left Right Fig. 1 Findings from dominl ultrsonogrphy, CT, nd MRI. Adominl ultrsonogrphy shows n extensive spe-oupying lesion in the right loe of the liver, 15 m in dimeter. Left photo shows murl nodule, nd right photo shows heterogeneous internl omponent inluding hemorrhge nd hypervsulrity (lk rrow). Adominl ontrst-enhned omputed tomogrphy shows ysti mss mesuring m in the right loe of the liver with n enhned murl nodule (white rrow). d Adominl mgneti resonne imging (MRI) shows hyperintense omponent on T2-weighted imging omptile with the hemorrhgi re

3 Minmi et l. Surgil Cse Reports (2017) 3:66 Pge 3 of 5 Fig. 2 Findings of FDG-PET. The fusion imge of FDG-PET shows norml umultion in the liver nd lower domen (lk rrows). Cross-setion of the upper domen indites norml umultion in the murl nodule in the liver (white rrow). Cross-setion of the lower domen indites norml umultion of the omentl tumor (white rrow) history of sestos exposure. Among the remining 10 ses, 8 ses hd no history of sestos exposure nd 3 were not evluted. One se hd history of virl heptitis, nd 9 ses did not. Men tumor size ws 12.2 m (rnge, m), nd our se showed the third lrgest mss. Gross finding of the tumor ws ysti mss whih is not ommon, nd there ws no report of se with ysti tumor ever. This tumor ontined hemorrhgi fluid; therefore, we thought tht the tumor might hve led nd formed ysti mss. In 10 ses, the tumor rose in the right loe, s in our se, nd only one tumor rose in the left loe. Surgery ws performed in 10 ses, nd only 1 se reeived est supportive re, due to systemi lymph node swelling nd rupture of the tumor [2]. Pthologilly, mlignnt mesotheliom is lssifile into three sutypes: epithelioid, sromtoid, nd iphsi. The epithelioid type is the most ommon type, nd tuuloppillry or solid vritions n e seen in the tumor [3]. The tumor in our se ws lso dignosed s epithelioid type, ut showed typil findings suh s solid nd tuulr, ysti, or spindle omponents in the tumor, lerly prtitioned from norml liver tissue y firous psule. The tumor showed prtil hyliniztion, ut no nerosis. Typil immunohistohemil fetures re positive results for lretinin, vimentin, ytokertin, WT-1, nd D2-40 nd negtive results for CD34, CEA, AFP, nd Ber-EP4, s seen in our se [3]. In terms of tumor prolifertive tivity, Ki-67 index in the typil mlignnt mesotheliom is 15 20% [11], ut ws 5 6% in our se. The tumor ws therefore Fig. 3 Gross findings of speimens. The hepti tumor represents mssive ysti tumor ontining hemorrhgi fluid nd mesuring m. The ut surfes of the tumor show muinous or hemorrhgi, rownish or yellowish multinodulr tumor in the extrysti wll re (white rrow). The ut surfes of the omentl tumors indite multiple solid, rownish, nodulr tumors mesuring nd m (white rrows)

4 Minmi et l. Surgil Cse Reports (2017) 3:66 Pge 4 of 5 d e f Fig. 4 Histologil findings of tumors. Histologil speimen of liver tumor (hemtoxylin nd eosin (HE), 200) shows epithelioid-type mesotheliom ells with tuulr omponents. Mesotheliom ells with ysti omponents (HE, 200). Immunohistohemil stining for lretinin shows positive tumor ells ( 200). d Immunohistohemil stining for CEA shows negtive tumor ells ( 200). e Immunohistohemil stining for HepPer1 shows negtive tumor ells ( 200). f Immunohistohemil stining for Ki-67, mrker of tumor prolifertion, shows positive tumor ells (rown nulei indited with lk rrows). Ki-67 index is 5 6% dignosed s low-grde mlignnt tumor, nd metstsis ws onsidered unlikely, lthough primry mlignnt omentl mesotheliom is lso rre disese. We found only 2 se reports of mlignnt mesotheliom of the omentum reserhing with key words of greter omentum, mlignnt, nd mesotheliom [13, 14]. It ws diffiult to distinguish multiple tumor from metstti omentl tumor in our se. Multiple mlignnt mesothelioms nd metstsis of low-grde mlignnt mesotheliom re oth unlikely. The omentl tumor ells were positive for AE1/AE3, CK19, CK7, EMA, C D10, nd Tle 1 Summry of hepti mesotheliom Ctegories Dt (n = 12) Men ge (rnge) 58.4 yers (41 68 yers) Mle/femle 6/6 Asestos exposure (+/ /NE) 1/8/3 Virl heptitis (+/ /NE) 1 (C type)/9/2 Men tumor size (rnge) 12.2 m ( m) Lotion (Right/left/NE) 10/1/1 Tretment (Surgery/BSC/NE) 10/1/1 Pthologil type (Epithelioid/sromtoid/iphsi) 9/0/3 Relpse (+/ /NE) 2 (Lymph node)/5/5 NE not evluted; BSC est supportive re lretinin, prtly for CK5/6, D2-40, nd vimentin, nd negtive for HepPer1, hromogrnin A, synptophysin, CEA, inhiin, MUC1, meln A, HMB45, CA19-9, ER, PgR, CD34, l-2, nd β-tenin tht ws sme findings with hepti tumor. However, oth tumors hve firous psule without invsion of tumor ells. Moreover, oth tumors hd lower proliferted tivity nd onsidered to e low-grde mlignnt tumor. Bsing on these pthologil findings, we should dignose the tumors s multiple mesothelioms, lthough we re not le to deny possiility of dissemintion. Conerning out the outome, lymph node relpse hs only een reported in 2 ses, nd they were live t 2 or 36 months fter relpse without hemtogenous metstti disese [6, 9]. Our ptient remins live s of 6 months fter surgery without relpse. Conlusions We presented the se of 36-yer-old womn with multiple mlignnt mesotheliom of the liver nd greter omentum with review of the litertures. Funding We delre tht eh uthor reeived no funding for this study. Avilility of dt nd mterils The dtsets supporting the onlusions re inluded within the rtile. Authors ontriutions KM, HO, nd KH prtiipted in the surgery, olleted the dt nd onsent, nd drfted the mnusript. SM performed the pthologil exmintions nd prtiipted in drfting the mnusript.yo,ts,km,ys,ndmsprtiiptedin the surgery. IK dignosed the rdiologil findings nd prtiipted in the drfting of the mnusript. YB performed the physil dignosis. SN prtiipted in the design, oordintion, nd drfting of the mnusript. All uthors red nd pproved the finl mnusript.

5 Minmi et l. Surgil Cse Reports (2017) 3:66 Pge 5 of 5 Competing interests The uthors delre tht they hve no ompeting interests. Consent for pulition The ptient onsented to the pulition of the fetures of her se, nd her identity hs een proteted. Ethis pprovl nd onsent to prtiipte Not pplile. Pulisher s Note Springer Nture remins neutrl with regrd to jurisditionl lims in pulished mps nd institutionl ffilitions. Author detils 1 Deprtment of Surgery, Kgoshim Kouseiren Hospitl, Tenpozn 22-25, Kgoshim , Jpn. 2 Deprtment of Pthology, Kgoshim Kouseiren Hospitl, Tenpozn 22-25, Kgoshim , Jpn. 3 Deprtment of Rdiology, Kgoshim Kouseiren Hospitl, Tenpozn 22-25, Kgoshim , Jpn. 4 Deprtment of Digestive Surgery, Brest nd Thyroid Surgery, Grdute Shool of Medil Sienes, Kgoshim University, Skurgok , Kgoshim , Jpn. Reeived: 26 Ferury 2017 Aepted: 3 My 2017 Referenes 1. Zellos L, Christini DC. Epidemiology, iologi ehvior, nd nturl history of mesotheliom. Thor Surg Clin. 2004;14: Ingki N, Kit K, Tmki T, Shimizu T, Nomur S. Primry intrhepti mlignnt mesotheliom with multiple lymphdenopthies due to nontuerulous myoteri: se report nd review of the literture. Onol Lett. 2013;6: Serter A, Buyukpinrsili N, Krtepe O, Koko E. An unusul liver mss: primry mlignnt mesotheliom of the liver: CT nd MRI findings nd literture review. Jpn J Rdiol. 2015;33: Imur J, Ihikw K, Tked J, Iwski Y, Tomit S, Kuot K, et l. Lolized mlignnt mesotheliom of the epithelil type ourring s primry hepti neoplsm: se report with review of the literture. APMIS. 2002; 110: Leonrdou P, Semelk RC, Knemtsu M, Brg L, Woosley JT. Primry mlignnt mesotheliom of the liver: MR imging findings. Mgn Reson Imging. 2003;21: Gutgemnn I, Stndop J, Fisher HP. Primry intrhepti mlignnt mesotheliom of epithelioid type. Virhows Arh. 2006;448: Kim DS, Lee SG, Jun SY, Kim KW, H TY, Kim KK. Primry mlignnt mesotheliom developed in liver. Heptogstroenterology. 2008;55: Sski M, Arki I, Ysui T, Kinoshit M, Ittsu K, Nojim T, et l. Primry lolized mlignnt iphsi mesotheliom of the liver in ptient with sestosis. World J Gstroenterol. 2009;15: Buhholz BM, Gutgemnn I, Fisher HP, Gorshluter M, Turler A, Klff JC, et l. Lymph node dissetion in primry intrhepti mlignnt mesotheliom: se report nd implitions for dignosis nd therpy. Lngeneks Arh Surg. 2009;394: Dong A, Dong H, Zuo C. Multiple primry hepti mlignnt mesothelioms mimiking ystdenorinoms on enhned CT nd FDG PET/CT. Clin Nul Med. 2014;39: Perysinkis I, Nixon AM, Spyridkis I, Kkiopoulos G, Zorzos C, Mrgris I. Primry intrhepti mlignnt epithelioid mesotheliom. Int J Surg Cse Rep. 2014;5: Hji Ali R, Khlife M, El Nounou G, Zuhri Yfi R, Nssr H, Aidie Z, et l. Gint primry mlignnt mesotheliom of the liver: se report. Int J Surg Cse Rep. 2017;30: Shin MK, Lee OJ, H CY, Min HJ, Kim TH. Mlignnt mesotheliom of the greter omentum mimiking omentl infrtion: se report. World J Gstroenterol. 2009;15: Liu YC, Kuo YL, Yu CP, Wu HS, Yu JC, Chen CJ, et l. Primry mlignnt mesotheliom of the greter omentum: report of se. Surg Tody. 2004;34(9): Sumit your mnusript to journl nd enefit from: 7 Convenient online sumission 7 Rigorous peer review 7 Immedite pulition on eptne 7 Open ess: rtiles freely ville online 7 High visiility within the field 7 Retining the opyright to your rtile Sumit your next mnusript t 7 springeropen.om