A rare case of a spontaneously ruptured secondary hepatic malignant lymphoma

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1 Oshit et l. Surgicl Cse Reports (2018) 4:44 CASE REPORT Open Access A rre cse of spontneously ruptured secondry heptic mlignnt lymphom Ko Oshit 1, Toshiyuki Itmoto 1,3*, Akihiko Oshit 1,3, Hideki Nkhr 1 nd Tkshi Nishisk 2 Astrct Bckground: Although secondry liver involvement of the lymphom is common nd occurs in 50% of ptients with non-hodgkin s lymphom, liver tumor rupture in mlignnt lymphom is extremely rre. We report cse of spontneously ruptured secondry liver involvement of non-hodgkin s lymphom tht ws successfully treted with trnsctheter rteril emoliztion (TAE) to otin hemostsis, nd susequent heptectomy nd systemic chemotherpy. To the est of our knowledge, this is only the second reported cse of ruptured heptic lymphom. Cse presenttion: A 74-yer-old mn with sudden-onset right shoulder nd upper qudrnt pin ws trnsferred to our hospitl vi n mulnce. His vitl signs were stle. Contrst-enhnced computed tomogrphy showed lrge hypo-enhncing tumor (94 81 mm) in the posterior segment of the liver, with intrtumorl extrvstion nd mm dughter tumor in segment 5 of the liver. Hemoperitoneum due to rupture of heptocellulr crcinom with intrtumorl hemorrhge ws suspected, lthough the serum lph-fetoprotein nd protein induced y vitmin K sence-ii levels were within norml rnge. TAE ws used for hemostsis. Extended posterior segmentectomy including tumor resection in segment 5 ws performed on dy 23 fter emoliztion. The postopertive course ws uneventful. Pthologicl exmintion of the resected specimens reveled tht the ruptured tumor ws diffuse lrge B- cell lymphom. Postopertive fluorodeoxyglucose positron emission tomogrphy-computed tomogrphy showed uptke in the left protid glnd, pncres, nd thorcic verter. Bsed on these findings, the finl dignosis ws ruptured secondry heptic mlignnt lymphom. Complete remission ws chieved with chemotherpy. He remins live 30 months fter heptectomy without evidence of relpse. Conclusions: We report the first cse descriing heptic tumor rupture s the first presenttion of primry or secondry heptic mlignnt lymphom. The ptient ws successfully treted with TAE, heptectomy, nd susequent systemic chemotherpy for non-hodgkin s lymphom. Keywords: Heptic mlignnt lymphom, Spontneous rupture, Hemoperitoneum, Trnsctheter rteril emoliztion, Heptectomy Bckground The liver is n orgn tht cn either e involved in widespred lymphom or rrely s primry site of lymphom. Secondry liver involvement of the lymphom is common nd occurs in 50% of ptients with non- Hodgkin s lymphom. Moreover, the liver is the third * Correspondence: t-itmoto@hph.pref.hiroshim.jp 1 Deprtment of Gstroenterologicl Surgery, Hiroshim Prefecturl Hospitl, Ujin-knd, Minmi-ku, Hiroshim, Jpn 3 Deprtment of Gstroenterologicl nd Trnsplnt Surgery, Applied Life Sciences, Institute of Biomedicl nd Helth Sciences, Hiroshim University, Hiroshim, Jpn Full list of uthor informtion is ville t the end of the rticle most common dominl orgn with lymphom involvement, following the spleen nd gstrointestinl trct [1, 2]. The most common secondry heptic lymphom is non-hodgkin s lymphom [3]. Diffuse lrge B-cell lymphom is n ggressive non-hodgkin s lymphom with incresing incidence in elderly ptients. However, liver tumor rupture in ptients with mlignnt lymphom is extremely rre [4, 5]. To the est of our knowledge, this is only the second reported cse of ruptured heptic lymphom. Herein, we descrie ptient with spontneously ruptured secondry liver involvement of non-hodgkin s lymphom The Author(s) Open Access This rticle is distriuted under the terms of the Cretive Commons Attriution 4.0 Interntionl License ( which permits unrestricted use, distriution, nd reproduction in ny medium, provided you give pproprite credit to the originl uthor(s) nd the source, provide link to the Cretive Commons license, nd indicte if chnges were mde.

2 Oshit et l. Surgicl Cse Reports (2018) 4:44 Pge 2 of 6 successfully treted vi trnsctheter rteril emoliztion (TAE) to otin hemostsis nd vi susequent heptectomy nd systemic chemotherpy. Cse presenttion A 74-yer-old mn with sudden-onset right shoulder nd upper qudrnt pin cme to our hospitl vi n mulnce. His medicl history included chronic ostructive pulmonry disese nd ppendectomy. He reported no history of trum or flls. He hd not een prescried nticogulnts or ntipltelet gents. On physicl exmintion, he ws conscious nd lert, nd his lood pressure ws 109/65 mmhg; hert rte, 97 ets/min; nd respirtory rte, 20 reths/min. He complined of mild dominl distension nd tenderness on the right upper qudrnt re. Lortory exmintion showed white lood cells of 15,700/μL; hemogloin, 11.3 g/ dl, pltelet count, 236,000 /µl; sprtte minotrnsferse (AST), 57 U/L; lnine mino trnsferse (ALT), 43 U/L; serum level of lctte dehydrogense, 454 U/L; lkline phosphtse, 218 IU/L; nd totl-iliruin, 0.5 mg/dl. The results of cogultion tests were norml. Serum lph-fetoprotein nd protein induced y vitmin K sence-ii levels were within norml rnge (5 ng/ml nd 26 mau/ml, respectively). Heptitis B virus surfce ntigen nd heptitis C virus ntiody were negtive. Contrst-enhnced computed tomogrphy (CT) showed lrge hypo-enhncing tumor (94 81 mm) in the posterior segment of the liver with intrtumorl extrvstion nd mm dughter tumor in segment 5 (S5) of the liver (Fig. 1, ). Hemoperitoneum due to rupture of heptocellulr crcinom (HCC) with intrtumorl hemorrhge ws suspected, nd TAE ws selected for otining hemostsis. Selective heptic rteriogrm from the right posterior heptic rtery reveled tht the rteril rnches were stretching with peripherl enhncement of the tumor. No extrvstion of contrst medium could e detected during the procedure (Fig. 2). The right posterior heptic rtery ws emolized with geltin sponge (Fig. 2). The serum AST nd ALT levels, which were 358 nd 327 IU/L respectively, peked on the second dy nd improved within the norml rnge on the 12th dy fter TAE. The retention rte of indocynine green t 15 min ontheninthdyftertaews6.4%. Extended posterior segmentectomy (resection of S6 + S7) including removl of tumor in S5 of the liver ws performed on the 23rd dy fter TAE. Sustntil clots nd old lood were found in the dominl cvity. A tumor in the posterior segment dhered to the rightdiphrgm,ndprtofthetumorcpsulehd een ruptured. Intropertive echogrm nd plption reveled n incidentl tumor of 1.2 cm in dimeter in S3 of the liver. Additionl procedure included prtil resection of S3 of the liver. The resected specimen reveled tht the ruptured tumor in the posterior segment ws mm in size, nd the mjority of the tumor ws necrotized due to TAE (Fig. 3). c Fig. 1 Contrst-enhnced computed tomogrphy scn showed hypo-enhncing tumor (98 81 mm) in the posterior segment of the liver with intrtumorl extrvstion on rteril phse () nd on delyed phse (). A dughter tumor (12 10 mm) in segment 5 of the liver ws lso detected (c)

3 Oshit et l. Surgicl Cse Reports (2018) 4:44 Pge 3 of 6 Fig. 2 Trnsctheter rteril emoliztion ws performed. Selective heptic ngiogrm reveled tht the rnches of the right posterior heptic rtery were stretching with peripherl enhncement of the liver with no extrvstion (). The right posterior heptic rtery ws emolized with geltin sponge () Histologic exmintions reveled tht vile lesions were composed of diffusely proliferted typicl lymphoid cells (Fig. 4). Immunohistochemicl stining of these tumor cells ws negtive for heptocyte mrker, diffusely positive for CD20 nd CD79, nd foclly positive for Bcl- 2 (Fig. 4 d). Histologicl exmintion lso reveled mssive cogultive necrosis in the centrl prt of the tumor (Fig. 5). The Verhoeff-Vn Gieson stining of the re reveled reltively lrge rtery penetrting the necrotic tissue (Fig. 5). Approximtely 90% of the tumor cells were positive for Ki-67. These findings confirmed non-hodgkin s diffuse lrge B-cell lymphom. The serum interleukin-2 receptor level mesured fter identifiction of the pthologicl findings from the Fig. 3 Specimen of the resected posterior segment (S6/7) including tumor in S5 of the liver. The mjority of ruptured tumor ws necrotized

4 Oshit et l. Surgicl Cse Reports (2018) 4:44 Pge 4 of 6 c d Fig. 4 Hemtoxylin nd eosin stining ( 200 mgnifiction) demonstrted tht vile lesions in the tumor were composed of diffusely proliferted typicl lymphoid cells (). Immunohistochemicl stining ( 200 mgnifiction) reveled tht the tumor cells were diffusely positive for CD20 () nd CD79 (c) nd foclly positive for Bcl-2 (d) resected specimens ws 893 U/L. The postopertive course ws uneventful, nd he ws dischrged from the hospitl on postopertive dy 13. Fluorodeoxyglucose positron emission tomogrphycomputed tomogrphy (PET-CT) performed 1 month fter opertion showed n uptke in the left protid glnd, pncres, remnnt liver, nd the thorcic vertere. For systemic non-hodgkin s lymphom, the ptient received eight cycles of rituxim, cyclophosphmide, doxoruicin, vincristine, nd prednisolone regimen nd chieved complete remission, which hs een mintined to dte for 30 months. Discussion Rupture of liver tumor occsionlly occurs, nd the ssocited intrperitonel hemorrhge is potentilly lifethretening. The most common symptom of ruptured liver tumor is sudden onset of dominl pin with shock. HCC is the most common cuse of spontneous hemorrhge with 10% of these cses presenting with dominl leeding [6, 7]. However, heptic tumor rupture in ptients with mlignnt lymphom is extremely rre [4, 5]. Tsutni et l. first reported cse of ruptured mlignnt lymphom in 1999 [4]. Following initil tretment with chemotherpy nd irrdition for non-hodgkin s lymphom (diffuse lrge B-cell lymphom) of the nterior chest wll nd pr-ortic lymph nodes, disese recurrences occurred in the liver nd verter. When recurrent multifocl liver tumors decresed in size fter slvge chemotherpy including irinotecn, the ptient died of intrperitonel leeding. Autopsy reveled ruptured lymphoms of the liver. It ws speculted tht mrked nti-tumor effect reducing the tumor size might hve cused severe necrosis nd susequent rupture of the liver tumors. Fig. 5 Hemtoxylin nd eosin stining ( 40 mgnifiction) showed mssive cogultion in the centrl prt of the tumor (). Verhoeff-Vn Gieson stining ( 40 mgnifiction) of the re reveled reltively lrge rtery penetrting the necrotic tissue ()

5 Oshit et l. Surgicl Cse Reports (2018) 4:44 Pge 5 of 6 Our cse presented herein is the second report descriing heptic tumor rupture nd hemoperitoneum tht occurred in mlignnt lymphom. The ptient hs ruptured heptic tumor treted successfully vi TAE followed y heptectomy. The clinicl dignosis ws HCC rupture. Pthologicl exmintion of the resected specimens reveled tht the ruptured tumor ws non- Hodgkin s lymphom. Postopertive PET-CT reveled tht the ptient hd systemic disese of non-hodgkin s lymphom. In non-hodgkin s lymphom, liver involvement is oserved in s much s 50% of utopsy cses. However, tumor rupture nd hemoperitoneum hve never een descried s first presenttion of primry or secondry heptic mlignnt lymphom [8, 9]. This is the first report descriing such cse. Tsutni et l. suggested tht one of the resons for the low incidence of rupture of mlignnt lymphom might e its hypo-vsculrity [4]. Mjority of heptic lymphom lesions demonstrte miniml to no enhncement on ll the phses of contrst-enhnced CT. Enhncement, when present, is chrcteristiclly less thn the surrounding heptic prenchym [10, 11]. However, rteril chnnels cn often e seen coursing through the hypo-enhncing tumor [2]. The other pttern on contrst-enhnced CT is trget-like ppernce, tht is, peripherl enhncement with centrl non-enhncement, mimicking intrheptic cholngiocrcinom or metsttic liver cncer [11, 12]. In our cse, ruptured tumor ws hypo-enhncing with extrvstion of contrst medium within the tumor on contrst-enhnced CT. Although the hypo-vsculrity is not likely to e the ruptured HCC, clinicl dignosis of ruptured HCC ws mde, suggesting tht the hypovsculrity might depend on the intrtumorl hemtom due to hemorrhge or spstic chnges of the feeding rteries fter leeding. The mechnism of spontneous rupture in HCC hs een discussed nd might most likely involve vrious fctors. Spontneous rupture tends to occur in lrge tumors [13], ut smll lesions locted t the surfce of liver lso leed nd rupture [14, 15]. In the mjority of cses, leeding is proly initited within the tumor. This leds to sudden increse in pressure within the tumor followed y ursting through the tumor surfce [5, 16]. Bleeding within the tumor cn e cused y disruption of frile feeding rtery or y centrl necrosis in rpidly growing HCC [5, 7, 13]. Centrl necrosis my led to disruption of frile feeding rtery [7]. Some investigtors hve demonstrted impirment of the vsculr integrity in ptients presenting with spontneous rupture of HCC [17]. The sudden ostruction of venous dringe cused y rpid tumor growth or tumor thromus cn result in congestion nd further necrosis of tumor tissue nd cn led to incresed intrtumorl pressure [14, 16]. Another mechnism of tumor rupture includes lcertion of superficil tumor s result of repeted respirtory movement, prticulrly for tumor locted under the diphrgm [16]. In the present rre cse of rupture of secondry heptic lymphom, systemic mlignnt lymphom ws dignosed s result of heptic tumor rupture. The cuse of the tumor rupture might include leeding from rteril chnnels coursing through the hypo-enhncing tumor, which ws demonstrted y the existence of intrtumorl extrvstion of contrst medium on contrstenhnced CT. The mechnisms of tumor rupture in the present cse of mlignnt lymphom might e the sme s those of HCC rupture resulting from initil leeding within the tumor due to impirment of the vsculr integrity nd the susequent sudden increse in pressure within the tumor followed y ursting through the tumor cpsule. Although clinicl dignosis efore tretment ws ruptured HCC nd the tretment strtegy pplied ws similr for HCC, the ddition of chemotherpy for mlignnt lymphom fter surgery contriuted to good ptient prognosis. Conclusions We report the first cse of heptic tumor rupture s the first presenttion of primry or secondry heptic mlignnt lymphom. The ptient ws successfully treted vi TAE, heptectomy, nd susequent systemic chemotherpy for non-hodgkin s lymphom. Arevitions CT: Computed tomogrphy; FDG: Fluorodeoxyglucose; HCC: Heptocellulr crcinom; PET-CT: Positron emission tomogrphy-computed tomogrphy; TAE: Trnsctheter rteril emoliztion Author s contriutions KO nd TI conceived nd designed the study nd drfted the mnuscript. Authors other thn KO nd TI contriuted to the collection, nlysis, nd interprettion of dt. HN nd TI prticipted in the tretment of the ptient. TN performed the pthologicl nlysis. All uthors red nd pproved the finl mnuscript. TI hs the overll responsiility nd gurntees the scientific integrity of the pper. Ethics pprovl nd consent to prticipte Not pplicle Consent for puliction Written informed consent ws otined from the ptient for puliction of this cse report nd ny ccompnying imges. Competing interests The uthors declre tht they hve no competing interests. Pulisher s Note Springer Nture remins neutrl with regrd to jurisdictionl clims in pulished mps nd institutionl ffilitions. Author detils 1 Deprtment of Gstroenterologicl Surgery, Hiroshim Prefecturl Hospitl, Ujin-knd, Minmi-ku, Hiroshim, Jpn. 2 Deprtment of Pthology Clinicl Lortory, Hiroshim Prefecturl Hospitl, Hiroshim, Jpn. 3 Deprtment of Gstroenterologicl nd Trnsplnt Surgery, Applied Life

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