Portal vein aneurysm associated with arterioportal fistula after hepatic anterior segmentectomy: Thought-provoking complication after hepatectomy
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1 Kimur et l. Surgicl Cse Reports (2018) 4:57 CASE REPORT Open Access Portl vein neurysm ssocited with rterioportl fistul fter heptic nterior segmentectomy: Thought-provoking compliction fter heptectomy Yusuke Kimur *, Tomohide Hori *, Tkfumi Mchimoto, Ttsuo Ito, Toshiyuki Ht, Yoshio Kdokw, Shigeru Kto, Diki Ysukw, Yuki Aisu, Yuichi Tkmtsu, Tku Kitno nd Tsunehiro Yoshimur Astrct Bckground: Few cses of postopertive rterioportl fistul (APF) hve een documented. APF fter heptectomy is very rre surgery-relted compliction. Cse presenttion: A 62-yer-old mn ws dignosed with heptocellulr crcinom in segments 5 nd 8, respectively. Anterior segmentectomy ws performed s curtive surgery. Ech rnch of the heptic rtery, portl vein, nd iliry duct for the nterior segment ws ligted together s the Glissonen undle. The ptient ws dischrged on postopertive dy 14. Three months lter, dynmic mgnetic resonnce imging showed n rterioportl fistul nd portl vein neurysm. Surprisingly, the ptient did not hve sutle symptoms. Although perfect ngiogrphic evlution could not e ensured, we performed ngiogrphy with susequent interventionl rdiology to void sudden rupture. Arteriogrphy ws immeditely performed to crete portogrm vi the APF from the stump of the nterior heptic rtery, nd portogrphy clerly reveled heptofugl portl vein flow. Portogrphy lso showed tht the stump of the nterior portl vein hd developed 40-mm-dimeter portl vein neurysm. Selective emoliztion of the nterior heptic rtery ws ccomplished in the whole length of the stump of the nterior heptic rtery, nd norml lood flow through the APF ws drsticlly reduced. The portl vein neurysm disppered, nd portl flow ws normlized. Dynmic computed tomogrphy fter emoliztion clerly demonstrted perfect interruption of the APF. The ptient mintined good helth therefter. Conclusions: Post-heptectomy APFs re very rre, nd some pper to e cryptogenic. Our thoughtprovoking cse my help to provide possile explntion of the cuses of post-heptectomy APF. Keywords: Shunt, Arterioportl fistul, Portl vein neurysm, Compliction, Heptectomy, Intervention rdiology * Correspondence: ykimur@tenriyorozu.jp; horitomo55office@yhoo.co.jp Yusuke Kimur nd Tomohide Hori contriuted eqully to this work. Deprtment of Digestive Surgery, Tenri Hospitl, 200 Mishim-cho, Tenri City, Nr Prefecture , Jpn 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 Kimur et l. Surgicl Cse Reports (2018) 4:57 Pge 2 of 6 c d Fig. 1 Dynmic CT findings efore surgery. Dynmic CT reveled two tumors (rrows) locted in, segment 8 nd c, d segment 5., c Findings in the erly phse., d Findings in the delyed phse Short description Arterioportl fistul (APF) is rre condition, nd some APFs pper to e cryptogenic. Few cses of postopertive APF hve een documented, nd APF fter heptectomy is very rre surgery-relted compliction. We herein presented thought-provoking cse of surgery-relted APF fter heptectomy. Our cse my e informtive with respect to explining the possile cuses of APF fter heptectomy. Bckground An rterioportl fistul (APF) or shunt is rre condition. An APF cuses portl hypertension [1] nd sometimes results in life-thretening events (e.g., liver filure, heptic encephlopthy, nd vricel leeding) [2 4] requiring surgicl tretment, interventionl rdiology (IVR), nd endovsculr therpy [2, 5]. The known etiologies of APF include trum, itrogenic cuses (e.g., iliry dringe, percutneous iopsy, nd rdiofrequency ltion), congenitl Fig. 2 RI findings enhnced y Gdolinium-ethoxyenzyl-diethylenetrimine pentcetic cid efore surgery. Enhnced MRI reveled two tumors (rrows) locted in segment 8 nd segment 5
3 Kimur et l. Surgicl Cse Reports (2018) 4:57 Pge 3 of 6 14], nd mssive leeding from vrices or neurysms will ffect the postopertive course fter heptectomy [13, 15]. APF is very rre compliction fter heptectomy [16], lthough cses of APF fter gstrectomy nd lproscopic cholecystectomy hve een documented [17, 18]. To the est of our knowledge, only one cse of APF fter heptectomy hs een reported [16]. We herein report thought-provoking cse of APF fter nterior segmentectomy. We lso discuss possile explntion for the cuse of APF in this cse. Fig. 3 Intropertive findings of FSTG. Anterior segmentectomy ws performed. Ech rnch of the heptic rtery, portl vein, nd iliry duct were ligted together s the Glissonen undle (so-clled FSTG) (rrow) disese, mlignnt tumors, nd splnchnic rtery neurysm rupture [1 4, 6 8]. Mjor heptectomy is currently fesile nd sfe therpeutic option for liver disese [9, 10], lthough some postopertive complictions re intrctle (e.g., iliry lekge, portl thromosis, nd refrctory scites) [11 13]. Ftl complictions hve een reported, such s liver filure, secondry portl hypertension, nd norml hemostsis [13, Cse presenttion A 62-yer-old mn with chronic heptitis C ws referred y his physicin to our hospitl for surgicl tretment of heptocellulr crcinom. Imging findings on enhnced computed tomogrphy (CT) nd dynmic mgnetic resonnce imging (MRI) reveled two tumors locted in segments 5 nd 8, respectively (Figs. 1 nd 2). Although the lph-fetoprotein level ws within the reference rnge, the serum level of prothromin induced y the sence of vitmin K or ntgonist-ii ws high (530 mau/ml). After preopertive evlution sed on three-dimensionl (3D) imging study, nterior segmentectomy ws performed. Ech rnch of the heptic rtery, portl vein, nd iliry duct for the nterior segment were ligted together s the Glissonen undle (so-clled, fully simultneous trnsection of the c d Fig. 4 Dynmic MRI findings 3 months fter surgery., Axil imges of dynmic MRI. c, d Coronl imges of dynmic MRI. An rterioportl fistul nd portl vein neurysm were incidentlly detected. Lyers of old nd sucute hemtoms were clerly oserved, nd these lyers surrounded the neurysm (rrows). We suspected pseudoneurysm sed on these MRI findings
4 Kimur et l. Surgicl Cse Reports (2018) 4:57 Pge 4 of 6 c d Fig. 5 Findings of ngiogrphy. Angiogrphy vi the celic rtery ws performed to crete portogrm vi the APF, nd portogrphy clerly reveled heptofugl flow of the portl vein (lue rrows). Portogrphy lso showed tht the stump of the nterior portl vein hd developed PVA with dimeter of 40 mm (ornge rrows). Selective ctheteriztion of the common heptic rtery clerly demonstrted the APF t the stump of the nterior rnches (red rrows). A definitive dignosis of PVA due to APF ws mde. An dequte length of APF to perform emolic therpy ws confirmed. c Selective emoliztion of the nterior heptic rtery ws ccomplished y plcement of severl titnium coils (yellow rrow). Blood flow through the APF ws drsticlly reduced. d Arteriogrphy vi the superior mesenteric rtery showed heptopetl portl flow (purple rrows) Glissonen pedicle [FSTG]) (Fig. 3). Perihilr FSTG involved trnsfixtion suture y using n sorle thred. The tumor in segment 8 ws in contct with the middle heptic vein. However, this tumor ws well-encpsulted, nd the tumor nd vein were esily dissectle. The ptient s postopertive course ws uneventful, nd he ws dischrged on postopertive dy 14. Three months lter, dynmic MRI ws performed to check for intrheptic recurrence, nd no imging findings of recurrence were oserved. However, n rterioportl fistul nd portl vein neurysm were incidentlly detected (Fig. 4). Lyers of old nd sucute hemtoms were clerly oserved, nd these lyers surrounded the neurysm. Surprisingly, the ptient did not hve sutle symptoms nd showed no episodes of pin, scites, liver dysfunction, or other normlities. We suspected pseudoneurysm t tht time. Although perfect ngiogrphic evlution could not e ensured, IVR ws needed to void sudden rupture nd possile deth. Therefore, we decided to ttempt IVR fter evlution of the vessels on dynmic CT, nd trnsctheter rteril emoliztion ws proposed therefter. First, ngiogrphy vi the celic rtery ws performed. Arteriogrphy ws susequently used to crete portogrm vi this APF, nd portogrphy clerly reveled heptofugl flow of the portl vein. Portogrphy lso showed tht the stump of the nterior portl vein hd developed portl vein neurysm (PVA) with dimeter of 40 mm (Fig. 5). Selective ctheteriztion of the common heptic rtery ws then performed. This rteriogrphy clerly demonstrted fistul etween the heptic rtery nd portl vein (i.e., APF) t the stump of the nterior rnches (Fig. 5). Bsed on these ngiogrphy findings, we definitively dignosed PVA due to n APF, not pseudoneurysm. Next, n dequte length of APF to perform emolic therpy ws confirmed to void ny occlusion nd disturnce t the ifurction of the right heptic rtery (Fig. 5). Selective emoliztion of the nterior heptic rtery ws then ccomplished y plcing severl titnium coils in the whole length of the stump of the nterior heptic rtery. Finlly, the flow of lood through the APF ws drsticlly reduced (Fig. 5c). Arteriogrphy vi the superior mesenteric rtery showed remrkle
5 Kimur et l. Surgicl Cse Reports (2018) 4:57 Pge 5 of 6 Fig. 6 Imging findings efore nd fter IVR., Imging findings efore IVR. Prior to IVR, the APF nd PVA were detected y dynmic CT nd 3D imges. c, d Imging findings fter IVR. Three dys fter emoliztion, c dynmic CT nd d 3D imges clerly demonstrted oth perfect interruption of the APF nd disppernce of the PVA restortion of portl venous flow, nd heptopetl portl flow ws clerly oserved (Fig. 5d). Complete closure of the APF could e estimted y dditionl expnsion of the metllic coils over time fter IVR. Dynmic CT nd 3D imges 3 dys fter emoliztion clerly demonstrted perfect interruption of the APF nd disppernce of the PVA (Fig. 6). Imging studies nd serum iomrkers showed no evidence of recurrence. At the time of this study, the ptient ws good in helth nd hd een reintegrted into society. Conclusions Cuses of APF include trum, itrogenic cuses (e.g., iliry dringe, percutneous iopsy, nd rdiofrequency ltion), congenitl disese, mlignnt tumors, nd splnchnic rtery neurysm rupture [1 4, 6 8]. Some APFs pper to e cryptogenic. Surgery-relted APFs re rre, lthough cses of APF fter gstrectomy nd lproscopic cholecystectomy hve een reported [17, 18]. To the est of our knowledge, however, only one cse of APF fter heptectomy hs een documented, nd this previous cse occurred in 3-month-old infnt fter right trisegmentectomy [16]. The infnt hd een successfully treted y super-selective emoliztion using titnium coils. His rtery, portl vein, nd ile duct were ligted, respectively. This pproch ws distinct from our pproch, FSTG. A simple question rose in the present cse: Is FSTG dngerous during heptectomy? FSTG is currently sfe nd reproducile heptectomy technique ecuse of its simplicity, nd this technique hs therefore ecome stndrd method during mjor heptectomy [9, 10]. In our institution, we lso employ perihilr FSTG during mjor heptectomy for heptocellulr crcinom, metsttic tumors, nd enign diseses. In the present cse, we understood tht it is difficult to discuss the mechnism of APF development. We considered tht our cse ws n gnogenic APF nd tht reltion etween perihilr FSTG nd resultnt APF ws lso uncler. Our procedures of perihilr FSTG involved trnsfixtion suture, nd we here speculted tht perihilr FSTG might hve een possile cuse of the APF. We lso speculted tht other possile cuses (e.g., technicl error) were oserved, nd recognized tht responsile cuse of our APF ws still oscure. We hd not experienced similr cses of APF fter heptectomy ccompnied y FSTG, lthough we found no reports of FSTG-relted APF. Although our ptient ws symptomtic, CT detected smll mount of scites. APFs, especilly those on the proximl side (e.g., intrheptic or perihilr Glissonen pedicle), often result in refrctory symptoms of portl hypertension (such s gstrointestinl leeding, scites,
6 Kimur et l. Surgicl Cse Reports (2018) 4:57 Pge 6 of 6 nd dirrhe) [6]. In the present cse, definitive dignosis of APF ws mde only 3 months fter heptectomy, nd we considered tht the reson why the ptient hd no symptoms ws the prompt dignosis of APF followed y dequte IVR. If prompt dignosis followed y dequte therpy hd filed, his portl hypertension would likely produce intrctle symptoms over time. A simple question rose in the present cse: Why symptoms of portl hypertension did not pper in this cse? We speculted one possile reson ws tht APF ws developed not t the min trunk level ut t the stump of nterior Glissonen pedicle which ws locted t reltively peripherl lesion compred with min trunk. Therpeutic strtegies for APFs include surgery (e.g., prtil heptectomy nd ligtion of the relted heptic rtery) nd IVR (e.g., trnsrteril emoliztion). Our ptient hd n dequte length etween the stump of the nterior heptic rtery nd the ifurction of the right heptic rtery, nd it ws considered suitle for trnsrteril emoliztion. We suggest tht ngiogrphy should e considered s the first-choice imging technique to elucidte the detils of APFs nd susequently determine the optiml therpy [19]. Glissonen pedicle trnsection (i.e., FSTG) is now routinely employed during heptectomy worldwide. We consider this mneuver to e very useful during mjor heptectomy. The occurrence of APF fter surgery is considered to e low [16 18]. However, once n APF hs developed, the ptient will experience poor clinicl course ccompnied y severe portl hypertension, nd this intrctle compliction requires surgicl or interventionl tretment. APF fter heptectomy is very rre, nd some APFs pper to e cryptogenic. Our thought-provoking cse my e informtive in terms of providing possile explntion of the cuses of APF fter heptectomy. Arevitions 3D: Three-dimensionl; APF: Arterioportl fistul; CT: Computed tomogrphy; FSTG: Fully simultneous trnsection of the Glissonen pedicle; IVR: Interventionl rdiology; MRI: Mgnetic resonnce imging; PVA: Portl vein neurysm Authors contriutions YK nd THo collected the dt. YK wrote n initil drft. THo nd TI revised the further drfts. YK nd THo contriuted eqully to this work. All uthors provided cdemic opinions nd helped to ssess importnt ppers. TY supervised this report. All uthors red nd pproved the finl mnuscript. Competing interest The uthors declre tht they hve no competing interests. Ethics pprovl nd consent to prticipte The report ws pproved y the Institutionl Review Bord of Tenri Hospitl. The ptient involved in this report provided his written informed consent uthorizing the use nd disclosure of his protected helth informtion. Pulisher s Note Springer Nture remins neutrl with regrd to jurisdictionl clims in pulished mps nd institutionl ffilitions. Received: 19 Ferury 2018 Accepted: 4 June 2018 References 1. Zhng DY, Weng SQ, Dong L, Shen XZ, Qu XD. Portl hypertension induced y congenitl heptic rterioportl fistul: report of four clinicl cses nd review of the literture. World J Gstroenterol. 2015;21: Guzmn EA, McChill LE, Rogers FB. Arterioportl fistuls: introduction of novel clssifiction with therpeutic implictions. J Gstrointest Surg. 2006; 10: Hori T, Ued M, Oike F, Ogur Y, Ogw K, Nguyen JH, et l. Grft loss nd poor outcomes fter living-donor liver trnsplnttion owing to rterioportl shunts cused y liver needle iopsies. Trnsplnt Proc. 2010; 42: Otoe Y, Hshimoto T, Shimizu Y, Nkmur T, Ymmori N, Hyshi S, et l. Formtion of ftl rterioportl fistul following needle liver iopsy in child with living-relted liver trnsplnt: report of cse. Surg Tody. 1995;25: Kumr A, Ahuj CK, Vys S, Klr N, Khndelwl N, Chwl Y, et l. Heptic rteriovenous fistule: role of interventionl rdiology. 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