Laparoscopic resection of idiopathic jejunal arteriovenous malformation after metallic coil embolization

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1 So et l. Surgicl Cse Reports (2018) 4:78 CASE REPORT Lproscopic resection of idiopthic jejunl rteriovenous mlformtion fter metllic coil emoliztion Mkiko So, Yoshiro Ittni * nd Yoshihru Ski, Kzutk Om, Shigeru Tsunod, Shigeo Hismori, Kyoichi Hshimoto Open Access Astrct Bckground: Arteriovenous mlformtions (AVM) developed in the smll intestine re rre, nd it is sometimes difficult to identify nd tret leeding from smll intestinl AVMs endoscopiclly ecuse of their locliztion. We present cse of jejunl AVM successfully treted with the comintion of metllic coil emoliztion nd lproscopic surgery. Cse presenttion: A 50-yer-old womn with history of repetitive gstrointestinl leeding ws dmitted to the hospitl. Selective ngiogrphy reveled jejunl AVM tht ws treted with metllic coil emoliztion. However, the lesion reled 3 months lter, nd it ws emolized gin with metllic coils. Considering the risk of releeding, we performed lproscopic resection of the jejunl AVM. Under lproscopy lone, it ws impossile to detect the lesion of the AVM. We used X-ry fluoroscopy intropertively to detect the metllic coils t the AVM. Prtil resection of the jejunum with the AVM ws performed followed y functionl end-to-end nstomosis. The ptient ws dischrged from the hospitl without ny complictions fter the surgery. Conclusions: The comintion of metllic coil emoliztion y ngiogrphy nd lproscopic surgery with X-ry fluoroscopy cn e effective for ptients with repetitive leeding from jejunl AVM. Keywords: Lproscopy, Jejunl AVM, Metllic coil emoliztion Bckground Gstrointestinl rteriovenous mlformtions (AVM) often cuse gstrointestinl leeding, ut sometimes they re difficult to dignose y endoscopy in the sence of ctive leeding. In prticulr, smll intestinl AVM cnnot e dignosed y either upper or lower endoscopy ecuse of its loction, even if it is ctively leeding. The dignosis of smll intestinl AVMs is usully mde y selective mesenteric rteriogrphy, demonstrting chrcteristic vsculr tufts nd very erly venous phse [1]. In 1986, Kndrp et l. reported the efficcy of coil emoliztion under ngiogrphyjusteforesurgeryto loclize intestinl AVMs [2]. Although n endoscopic exmintion is effective when the lesion is ccessile, selective mesenteric ngiogrphy is the stndrd * Correspondence: ittni@kuhp.kyoto-u.c.jp Deprtment of Surgery, Grdute School of Medicine, Kyoto University, Kyoto , Jpn exmintion to dignose, loclize, nd tret smll intestinl AVM. If the leeding is uncontrollle y either endoscopy or ngiogrphic emoliztion, surgicl resection of the intestine ffected is necessry. We present here rre cse of jejunl AVM successfully treted with the comintion of metllic coil emoliztion nd lproscopic surgery, nd reviewed cses of smll intestinl AVM resected lproscopiclly. Cse presenttion A 50-yer-old womn presented to our hospitl with hemtochezi nd nemi. 1 yer erlier, she experienced severe nemi (hemogloin 4.0 g/dl) tht ws treted with lood trnsfusion t nother hospitl. The dignosis t tht time ws hemorrhgic gstric ulcer, nd she ws treted with proton pump inhiitor. Contrst-enhnced dominl computed tomogrphy (CT) done just efore the first dministrtion to our 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.

So et l. Surgicl Cse Reports (2018) 4:78 Pge 2 of 5 hospitl showed multiple liver lesions of rterioportl nd portl venous shunts, hemngioms, nd lrge focl nodulr hyperplsi.

2 So et l. Surgicl Cse Reports (2018) 4:78 Pge 2 of 5 hospitl showed multiple liver lesions of rterioportl nd portl venous shunts, hemngioms, nd lrge focl nodulr hyperplsi. She hd hemtochezi nd nemi (hemogloin 7.0 g/dl) gin nd ws referred to our hospitl for further exmintion. Upper nd lower gstrointestinl endoscopies including doule-lloon enteroscopy did not revel ny leeding lesions in her esophgus, stomch, duodenum, proximl jejunum, colon, or rectum, lthough she hd grde 1 esophgel vrices. Angiogrphic exmintion reveled n AVM, with signs of extrvstion, t the jejunl rnch of the superior mesenteric rtery (SMA; Fig. 1). Three vs rect rnches of the jejunum t the AVM lesion were emolized with metllic coils to stop the leeding (Fig. 1). The ptient ws then dischrged from the hospitl without ny complictions. Three months fter the emoliztion, she experienced hemtochezi nd nemi gin nd ws dmitted to our hospitl. Repet ngiogrphy showed releeding from the sme AVM, nd n dditionl 3 vs rect rnches were treted with metllic coil emoliztion (Fig. 2). The coil emoliztion ws temporrily successful gin. However, ecuse of the risk of nother releeding from the sme AVM in ddition to the risk of necrosis of the coil-emolized jejunum, we considered resection of the ffected jejunum to e the optiml tretment nd recommended this to the ptient. Elective lproscopic surgery ws performed under generl nesthesi. Although initil investigtion under lproscopy lone filed to loclize the lesion, X-ry fluoroscopy showed cler imge of the metllic coils emolizing the AVM (Fig. 3). Susequently, the smll owel ws tken out through the umilicl incision, nd the metllic coils were confirmed y plption under direct vision. Prtil resection of the jejunum ws performed, followed y functionl end-to-end nstomosis using liner stplers (Fig. 3). Pthologicl exmintion reveled firous thickening of the vessels nd infiltrtion of inflmmtory cells in the mesentery, suggesting focl inflmmtion in response to the coil emoliztion (Fig. 4). There ws no necrotic intestine cused y the emoliztion (Fig. 4). She hd no complictions fter surgery nd ws dischrged within 1 week. She did not hve ny hemtochezi fter resection of the AVM during 8 months of follow-up. Discussion AVM represents n norml connection of rteries nd veins. It occurs mostly in the hed nd neck, including in the centrl nervous system, ut cn pper in ny loction in the ody. In the gstrointestinl trct, it cn cuse cute nd chronic leeding, which cn e ftl. A gstrointestinl AVM cn e dignosed y ngiogrphy, showing norml vessels s smll vsculr tuft, usully fed y single rtery [1]. These rteries demonstrte rpid filling, tortuous, dilted, nd opque terminl erry -like structures, ccompnied with erly filling of reltively enlrged drining veins [3]. An endoscopic pproch is effective for AVMs locted in the esophgus, stomch, duodenum, colon, or rectum [4]. On the other hnd, smll intestine AVMs re very difficult to rech y endoscopy nd there re few cse reports in which smll intestine endoscopies were used to detect them [5, 6]. In our cse, we did try smll intestine endoscopy, ut filed to detect ny leeding lesions. Fig. 1 Selective ngiogrphy of the SMA. Extrvstion (red circle) from the jejunl rnch of the SMA (red rrow) supplying the AVM is clerly oserved. Three vs rect rnches of the jejunl rnch emolized with metllic coils (red rrowheds), with no extrvstion from the AVM evident fter coil emoliztion

So et l. Surgicl Cse Reports (2018) 4:78 Pge 3 of 5 Fig. 2 Second selective ngiogrphy of SMA (red rrow). Extrvstion (red circle) detected t the sme AVM emolized during the first ngiogrphy.

Additionl metllic coils (red rrowheds) emolized to 3 vs rect rnches close to the previously emolized rteries, with no extrvstion evident fter emoliztion Once ptients need surgery for the tretment of

3 So et l. Surgicl Cse Reports (2018) 4:78 Pge 3 of 5 Fig. 2 Second selective ngiogrphy of SMA (red rrow). Extrvstion (red circle) detected t the sme AVM emolized during the first ngiogrphy. Red rrow indictes the jejunl rnch of the SMA. Additionl metllic coils (red rrowheds) emolized to 3 vs rect rnches close to the previously emolized rteries, with no extrvstion evident fter emoliztion Once ptients need surgery for the tretment of smll intestine AVMs, it cn e difficult to loclize the lesions during surgery. To overcome this prolem, severl intropertive techniques hve een reported. Evns et l. reported the effectiveness of mesuring intropertive mesenteric venous pressure nd PO2 [7]. In this pproch, the venous return from the AVM ws chrcterized y n elevted venous pressure nd PO2 levels compred with those from djcent norml intestines. The sme group lso reported the intropertive use of Doppler ultrsound to detect intestinl AVMs [8]. Defreyne et l. reported the intropertive use of methylene lue dye injection to visulize the loction of smll intestine AVM [9]. Recently, Ono et l. reported tht n intropertive indocynine green dye injection nd exmintion with fluorescent scope could loclize jejunl AVMs [10]. Although these techniques descried ove re useful in open surgery, they re sometimes difficult to e performed nd oserved during lproscopic surgery with limited view re on screen. As mentioned ove, coil emoliztion prior to surgery is lso useful for the locliztion of smll intestine AVMs ecuse the coil is plple intropertively in open surgery, nd detectle under X-ry fluoroscopy in oth open nd lproscopic surgery. In ddition to the useful technique for the locliztion intropertively, coil emoliztion cn lso e powerful method for treting cute leeding from the AVM. In our cse, we employed this method t the ptient s first presenttion t our hospitl nd were le to stop leeding from the AVM without ny ischemic chnges to the jejunum. Unfortuntely, however, the sme AVM reled 3 months lter nd ws successfully treted with second coil emoliztion. Although there seems to e no consensus out how mny vs rect rnches cn e emolized in cse of smll intestinl leeding, it is ovious tht n excess mount of emoliztion would result in cute or chronic ischemi of the intestine. In our cse, Fig. 3 Intropertive detection of metllic coils. X-ry fluoroscopy loclizing the metllic coils (red circle) emolized under the previous ngiogrphies. Forceps indicting one of the metllic coils fter tking out of the lesion detected under X-ry fluoroscopy

So et l. Surgicl Cse Reports (2018) 4:78 Pge 4 of 5 Fig. 4 Pthologicl exmintions of the resected tissue. Hemtoxylin nd eosin stining revels thickening of the vessels in the mesentery. Scle r, 200 μm.

4 So et l. Surgicl Cse Reports (2018) 4:78 Pge 4 of 5 Fig. 4 Pthologicl exmintions of the resected tissue. Hemtoxylin nd eosin stining revels thickening of the vessels in the mesentery. Scle r, 200 μm. Elstic-Msson stining revels collgenous firosis (green) of the vessel wlls. Inflmmtory cells such s lymphocytes, polymorphonucler cells, nd multinucleted gint cells re infiltrting round the vessels, suggesting focl inflmmtion ssocited with the coil emoliztion Tle 1 Previous reports of smll intestinl AVMs resected lproscopiclly Authors Title Intropertive detection of AVM Chung CS et l. [15] Kim SH et l. [11] Lee YH et l. [12] Shn B et l. [13] Fujii T et l. [5] Klmr P et l. [14] Mrtinez JC et l. [16] Fujikw T et l. [17] Muki M et l. [18] Otsuk S et l. [6] Ymmoto T et l. [19] Defreyne L et l. [9] Emergent single-lloon enteroscopy for overt leeding of smll intestinl vsculr mlformtion Vsculr mlformtions of the smll intestine mnifesting s chronic nemi: two peditric cses mnged y single-site umilicl lproscopic surgery A long-segmentl vsculr mlformtion in the smll owel presenting with gstrointestinl leeding in preschool-ged child Vsculr mlformtion s cuse of occult gstrointestinl leeding Arteriovenous mlformtion detected y smll owel endoscopy Lrge, segmentl, circulr vsculr mlformtion of the smll intestine (in femle toddler with hemtochezi): unusul presenttion in child Single incision lproscopic surgery pproch for oscure smll intestine leeding loclized y CT guided percutneous injection of methylene lue Successful resection of complicted leeding rteriovenous mlformtion of the jejunum in ptients strting dul-ntipltelet therpy just fter implnting drug-eluting coronry stent Intropertive fluoroscopic detection of n occult jejunl rteriovenous mlformtion Resection of rteriovenous mlformtion of the jejunum treted y lproscopy-ssisted surgery Resection of the rteriovenous mlformtion of the jejunum with the use of lproscopy-ssisted surgery comined with mrking coil Jejunl rteriovenous mlformtion, dignosed y ngiogrphy nd treted y emoliztion nd ctheter-guided surgery: cse report nd review of literture Article Yer Tttooing World J Gstroenterol 2018 Visile Int J Surg Cse Rep 2017 Visile Int J Rdiol 2016 Visile J Peditr Gstroenterol Nutr 2015 Not descried Cse Rep Gstroenterol 2014 Visile BMC Peditr 2014 Methylene lue Int J Surg Cse Rep 2014 Clip BMJ Cse Rep 2012 Methylene lue nd coil J Lproendosc Adv Surg Tech 2006 Intestinl endoscopy J Jpn Surg Assoc 2002 Coil Jpn J Gstroenterol Surg 1999 Methylene lue Adom Imging 1998

5 So et l. Surgicl Cse Reports (2018) 4:78 Pge 5 of 5 the ptient did not show ny cute complictions fter the second emoliztion, lthough totl of six vs rect rnches were emolized. There re 13 cses (12 reports) of smll intestinl AVM resected under lproscopic surgery (Tle 1). Among them, five cses were peditric, suggesting tht they were congenitl nd reltively lrge, nd the AVMs were directly visile intropertively [11 14]. As for other eight dult cses, they used tools descried ove for intropertive detection during lproscopic surgery in most of the cses [6, 9, 15 19]. If the AVMs were ccessile y doule-lloon enteroscopy, it would e powerful technique for locliztion of smll intestinl AVMs either y clipping or tttooing, or direct oservtion intropertively. On the other hnd, if the AVMs could not e oserved y endoscope, ngiogrphic pproch must e essentil for locliztion. Although intropertive injection of methylene lue could visulize the lesion of AVM, it needs ctheteriztion during opertion, which requires dditionl ptient cre in the opertion room. On the other hnd, emolized coils cn e left in the rnches of mesenteric rtery close to AVM for oth tretment nd locliztion purpose fter ngiogrphy, which gives us time to prepre the est preopertive mngement for the ptients. Conclusions We report rre cse of jejunl AVM treted with metllic coil emoliztion followed y lproscopic resection. This cse suggests the effectiveness of the comintion of metllic coil emoliztion nd lproscopic surgery for ptients with repetitive leeding from jejunl AVM. Arevitions AVM: Arteriovenous mlformtion; CT: Computed tomogrphy; SMA: Superior mesenteric rtery Acknowledgements We thnk ThinkSCIENCE for drft of this mnuscript. Funding No funding ws received for this cse report. Avilility of dt nd mterils Dt shring not pplicle to this rticle s no dtsets were generted or nlyzed during the current study. Authors contriutions MS nd YI wrote the mnuscript. KO supervised the cse. All other uthors reviewed the mnuscript. All uthors red nd pproved the finl mnuscript. Ethics pprovl nd consent to prticipte Not pplicle Consent for puliction Written informed consent ws otined from the ptient for the puliction of this report. Competing interests No potentil conflicts of interest exist. Pulisher s Note Springer Nture remins neutrl with regrd to jurisdictionl clims in pulished mps nd institutionl ffilitions. Received: 1 My 2018 Accepted: 11 July 2018 References 1. Cvett CM, Sely JH, Hmilton JL, Willimson JW. Arteriovenous mlformtion in chronic gstrointestinl leeding. Annls of Surg. 1977;185: Kndrp K, Fellows KE, Erklis A, Flores A. Solitry ilel rteriovenous mlformtion: preopertive locliztion y coil emoliztion. Am J Roentgenol. 1986;146: Crwford ES, Roehm JO, McGvrn MH. Jejunoilel rteriovenous mlformtion: locliztion for resection y segmentl owel stining techniques. Ann Surg. 1980;19: Hott M, Ymmoto K, Cho K, Tko Y, Fukuok T, Uchid E. Stomch rteriovenous mlformtion resected y lprocopy-ssisted surgery: cse report. Asin J Endosc Surg. 2015;9: Fujii T, Morit H, Sutoh T, Tkd T, Tsutsumi S, Kuwno H. Arteriovenous mlformtion detected y smll owel endoscopy. Cse Rep Gstroenterol. 2014;8: Otsuk S, Fuchimoto S, Oski T, Inoue F, Skt T, Miyoshi K. Resection of rteriovenous mlformtion of the jejunum treted y lproscopy-ssisted surgery. J Jpn Surg Assoc. 2002;63: Evns WE, O Dorisio TM, Molnr W, Mrtin EW Jr, Wooley CF, Coopermn M. Intropertive locliztion of intestinl rteriovenous mlformtion. Arch Surg. 1978;113: Coopermn M, Mrtin ES Jr, Evns WE, Crey LC. Use of Doppler ultrsound in intropertive locliztion intestinl rteriovenous mlformtion. Ann Surg. 1979;190: Defreyne L, Verstreten V, De Potter C, Pttyn P, De Vos M, Kunnen M. Jejunl rteriovenous mlformtion, dignosed y ngiogrphy nd treted y emoliztion nd ctheter-guided surgery: cse report nd review of literture. Adom Imging. 1998;23(2): Ono H, Kusno M, Kwmt F, Dnjo Y, Kwkmi M, Ngshim K, Nishihr H. Intropertive locliztion of rteriovenous mlformtion of jejunum with comined use of ngiogrphic methods nd indocynine green injection: report of new technique. Int J Surg Cse Rep. 2016;29: Kim SH, Cho YH, Kim HY. Vsculr mlformtions of the smll intestine mnifesting s chronic nemi: two peditric cses mnged y single-site umilicl lproscopic surgery. Int J Surg Cse Rep. 2017;31: Lee YJ, Hwng JY, Cho YH, Kim YW, Kim TU, Shin DH. A long-segmentl vsculr mlformtion in the smll owel presenting with gstrointestinl leeding in preschool-ged child. Int J Rdiol. 2016;13:e Shn B, Blinmn TA, Chill AM, Bhtti TR, Mqool A, Mmul P, Anupindi SA. Vsculr mlformtion s cuse of occult gstrointestinl leeding. J Peditr Gstroenterol Nutr. 2015;60:e Klmr P, Petnehzy T, Wieβpeiner U, Beer M, Huer AC, Till H, Riccon M. Lrge, segmentl, circulr vsculr mlformtion of the smll intestine (in femle toddler with hemtochezi): unusul presenttion in child. BMC Peditr. 2014;14: Chung CS, Chen KC, Chou YH, Chen KH. Emergent single-lloon enteroscopy for overt leeding of smll intestinl vsculr mlformtion. World J Gstroenterol. 2018;24: Mrtinez JC, Thoms JL, Lukszczyk JJ. Single incision lproscopic surgery pproch for oscure smll intestine leeding loclized y CT guided percutneous injection of methylene lue. Int J Surg Cse Rep. 2014;5: Fujikw T, Mekw H, Shirishi K, Tnk A. Successful resection of complicted leeding rteriovenous mlformtion of the jejunum in ptients strting dul-ntipltelet therpy just fter implnting drugeluting coronry stent. BMJ Cse Rep. 2012;10: Muki M, Iusuki K, Gotoh T, Ymmoto A, Nkshim K, Mutoh M, Tniguchi S, Kwno M, Kihr Y, Kog K. Intropertive fluoroscopic detection of n occult jejunl rteriovenous mlformtion. J Lproendosc Adv Surg Tech. 2006;16: Ymmoto T, Kwchi S, Kwhr H, Hmy M, Skuri T, Inoue S, Tkym S, Hr T. Resection of the rteriovenous mlformtion of the jejunum with the use of lproscopy-ssisted surgery comined with mrking coil. Jpn J Gstroenterol Surg. 1999;32:

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