Rev. Bras. Anestesiol. vol.66 número3

RevBrasAnestesiol.2016;66(3):318---320

REVISTA

BRASILEIRA

DE

ANESTESIOLOGIA

www.sba.com.br

CLINICAL

INFORMATION

Aberrant

right

subclavian

artery-esophageal

fistula:

massive

upper

gastrointestinal

hemorrhage

secondary

to

prolonged

intubation

Elsa

Oliveira

_,

_Margarida

_Anastácio,

_Anabela

_Marques

DepartmentofAnesthesiology,CentroHospitalareUniversitáriodeCoimbra,Coimbra,Portugal

Received24June2013;accepted25July2013 Availableonline23November2014

KEYWORDS

Aberrantright subclavianartery; Massivehemorrhage; Prolongedgastric intubation

Abstract Aberrantright subclavianartery-esophageal fistulaisarare but potentiallyfatal complication. Itmay be associated withprocedures, such astracheostomy andtracheal or esophagealintubation,andyieldsmassiveuppergastrointestinalbleedingdifficulttoidentify andtocontrol.

Ahighindexofsuspicionisessentialforearlydiagnosisandbetterprognosis.

Wereportararecaseofapatientwhosurvivedafteremergentsurgicalprocedurefor mas-siveuppergastrointestinalbleedingsecondarytoaberrantrightsubclavianartery-esophageal fistulaafterprolongedintubation.

©2014SociedadeBrasileiradeAnestesiologia.PublishedbyElsevier EditoraLtda.Allrights reserved.

PALAVRAS-CHAVE

Artériasubclávia direitaanômala; Hemorragiamacic¸a; Intubac¸ãogástrica prolongada

Fístuladeartériasubcláviadireitaanômalacomesôfago---hemorragiadigestivaalta macic¸asecundáriaaintubac¸ãogástricaprolongada

Resumo A fístulade artériasubclávia direita anômala como esôfago éuma complicac¸ão rara,mas potencialmente fatal.Pode estarassociada aprocedimentoscomo traqueostomia e intubac¸ão traqueal ou esofágica e originar hemorragia digestiva alta macic¸a, de difícil identificac¸ãoecontrole.

Umelevado índice de suspeic¸ão é essencial para o diagnóstico precoce e amelhoria do prognóstico.

∗_{Correspondingauthor.}

E-mail:elsacsoliveira@gmail.com(E.Oliveira). http://dx.doi.org/10.1016/j.bjane.2013.07.019

Aberrantrightsubclavianartery-esophagealfistula 319

Relatamos casoraro de doentequesobreviveuapós intervenc¸ãocirúrgica emergentepor hemorragiadigestivaaltamacic¸asecundáriaafístuladeartériasubcláviadireitaanômalacom esôfago,apósintubac¸ãogástricaprolongada.

©2014SociedadeBrasileira deAnestesiologia.PublicadoporElsevierEditoraLtda.Todosos direitosreservados.

Background

and

objectives

Aberrantrightsubclavianarteryisarelativelycommon con-genitalvascular anomaly(1---2%) incidence.1 _Mostpatients

havenosymptoms,whichmakesearlydetectiondifficult. The presence of aberrant right subclavian artery may have devastating consequences during procedures, such as tracheostomy, tracheal intubation, and surgical repair of thoracic aneurysms2 _{or during prolonged esophageal}

intubation.3 _{Gastric tubes can cause erosion of the}

esophageal wall resulting in fistula formation with the underlyingaberrantartery,causingmassivegastrointestinal bleedingthatisdifficulttoidentifyandcontrol.

Case

report

Amalepatient,20yearsold,wasadmittedtotheintensive careunitduetoseveretraumaticbraininjuries,thoracicand abdominaltrauma,andleftlowerlimbfracturesecondaryto acaraccident.Onadmission,thepatientwas hemodynam-icallystable,withaGlasgowComaScaleof4.Thepatient wasintubatedwitha7.5-cuffedtubeandmechanically ven-tilated.Healsohadanasogastrictubeonfreedrainageand urinarycatheter.

At 22 days of hospitalization, the patient remained sedated withmidazolamand intubatedfor airway protec-tionandmechanicalventilation,astherewerenoconditions forspontaneousventilation.Hewashemodynamicallystable andremainedwithurineoutputmonitoringandnasogastric tubeforenteralnutrition.

The patient developed suddenly upper gastrointestinal bleedingwithsignificant hematemesis.The gastroenterol-ogistcontactedforurgentendoscopyrevealed:‘‘Abundant amountofbloodandclotsthroughouttheesophagealpath. Bloodstreamseemstocomefromhighesophageallevel.The entiregastric cavityfilledwithmassiveclot andthesame happeningintheduodenallevel’’.Becauseoftheseverity ofbleedingandinability toidentifyitsorigin, thepatient underwentemergencysurgery.

Uponenteringtheoperatingroom,thepatienthad hypo-volemicshock,marked hypotension,intensepale mucous, and decreased hemoglobin values from 12.3gdL−1 _to

6.8gdL−1_.

Heunderwentbalancedanesthesiawithsevoflurane, fen-tanyl, and rocuronium for surgical intervention. Massive transfusionwasinitiatedwithrapidinfusionof colloidand crystalloidsolutionsandbloodproductsvia centralvenous

catheterplacedintherightfemoralveinandtwocatheters, 14Gand16G,inbothupperlimbs.

Duringexploratorylaparotomyandanteriorgastrostomy, itwasnotpossibletoidentifythesourceofbleeding. The gastroenterologistwasagaincontactedtoperformanother endoscopywhichidentified: ‘‘Ulcerwithcopious bleeding at the proximal esophagus level, at a distance of 20cm from the incisors’’. The option was a temporary closure of gastrostomy and abdomen, and an exploratory right posterolateralthoracotomy wasperformed, whichshowed arterio-esophageal fistula. Due to the need for differen-tiated surgical treatment for arterialfistula correction, a cardiothoracicsurgeonwascontacted.

Left thoracotomy wasperformed withidentification of controlledruptureofaberrantrightsubclavianartery.Right re-thoracotomywithligationof theaberrantright subcla-vianartery, esophagography,and abdominal closure were performed.

To controlmassivehemorrhage,18unitsof erythrocyte concentrate, 16units of fresh frozen plasma, two pools of platelets, prothrombin complex, and fibrinogen were administered,in additiontocolloid500mLandcrystalloid 1500mL.

Surgery lasted 8h. At the end of surgery, the patient wastransportedtothe intensivecare unit,intubatedand onmechanicalventilationmonitored.

The patientsurvived,evolvedfavorably andwas trans-ferred to the Department of Surgery. He was discharged one month and a half after this occurrence and was fol-lowedinoutpatientvisitsofsurgery,physicalmedicine,and rehabilitation.

Discussion

Themostcommonanomalyoftheaorticarchistheaberrant rightsubclavianartery,withanincidenceof1---2%.1_There

isapredominanceoffemales(65---72%).4_{Theaberrantright}

subclavianarteryformsanincompletevascularring, emerg-ing from the descending aorta and obliquely crosses the mediastinumtowardtherightarmpit.In80%ofthecases, it lies posterior to the esophagus (as in the clinical case described);in15%ofthecases,itliesbetweenthe esopha-gusandthetrachea;andin5%ofthecases,itliesanterior tothetrachea.5

320 E.Oliveiraetal.

It is extremely rare in the occurrence of bleeding by communicationbetweentheesophagusandanomalousright subclavianartery.Therearefewcasesreportedafter pro-longedesophagealintubation.3,7

The anatomicproximity totheesophagus and, eventu-ally,tothetrachearenderstheaberrantsubclavianartery vulnerabletoextrinsiccompression bygastric,trachealor vasculartubes.5

The relatively high incidence of this anomaly and the commonuseofgastrictubesinhospitalsincreasethe possi-bilityofthiscomplication.

Bleedingcausedbyesophagealfistulaandaberrantright subclavianarteryis manifestedbysudden massive hemor-rhagewithmassivefresharterialbloodhematemesis,often followedby an asymptomatic periodofvariableduration, andaperiodofsuddenandoftenfatalhemorrhage.3,8

Clin-icalsigns,suchasprecordialpainorsentinelhemorrhage, whicharepresent in 50% of thecasesof aortoesophageal fistula,arerarelypresentinthissituation.5

Facedwithmassivebleeding,thepriorityistoensureand protecttheairwayby trachealintubation. Inour patient, thisprocedure wasnot necessary,ashewasalready intu-bated and on mechanical ventilation, which may have improvedhisprognosis.Subsequently,thepriorityisto con-trolthebleeding.

Earlyperformanceofendoscopyisessentialfordiagnosis andexclusionofother potentialcausesofgastrointestinal bleeding. However, in most cases, massive bleeding pre-vents a conclusive endoscope examination. The diagnosis of thesource of bleeding is mostoften made intraopera-tivelyasalaparotomy and/orexploratorythoracotomy,as happenedinthiscase.Onlytherecognitionofthissituation willallow the establishment of immediate surgical treat-ment,withdecreasedhemodynamicchangesresultingfrom massivehemorrhage.

Patient’sprognosisdependsonearlydiagnosisand surgi-calrepair.5

Diagnostic difficulty in patients with massive hemor-rhagesecondarytofistulabetweenaberrantrightsubclavian

arteryandesophagusdeterminesthehighmortalityofthis complication.3

Ahigh indexof suspicionis essentialfor this condition earlydiagnosis.7

Thiscasereportaimstoraiseawarenessamong anesthe-siologistsforthepresenceofthiscongenitalanomaly,rarely identified, but potentially responsible for serious and/or fatalcomplicationsassociatedwithactsassimpleas naso-gastricortrachealtubeplacement.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

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2.WienbergerG,RandallPA,ParkerFB,etal.Involvementofan aberrantrightsubclavianarteryindissectionofthoracicaorta: diagnosticandtherapeuticimplications.AJR.1977;129:653---5. 3.MerchantFJ,NicholsRL,BombeckCT.Unusualcomplicationof

nasogastric esophagealintubation --- erosion into anaberrant rightsubclavianartery.JCardiovascSurg.1977;18:147---50. 4.EasterbrookJS.Identificationofaberrantrightsubclavianartery

on MR images of the cervical spine. J Magn Reson Imaging. 1992;2:507---9.

5.MillerRG,RobieDK,DavisSL,etal.Survivalafteraberrantright subclavianartery-esophagealfistula:casereportandliterature review.JVascSurg.1996;24:271---5.

6.NetoRC,FigueiraA,BelassaiE,etal.Hemorragiadigestivapor fístuladeartériasubcláviadireitaanômalacomesôfago.RevAss MedBras.1998;44:149---51.

7.Belkin RI, Keller FS, Everts EC, et al. Aberrant right sub-clavianartery --- esophageal fistula: a cause of overwhelming uppergastrointestinalhemorrage.CardiovascInterventRadiol. 1984;7:87---9.