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RevBrasAnestesiol.2015;65(6):525---528

REVISTA

BRASILEIRA

DE

ANESTESIOLOGIA

OfficialPublicationoftheBrazilianSocietyofAnesthesiology

www.sba.com.br

CLINICAL

INFORMATION

Anesthesia

for

ex

utero

intrapartum

treatment:

renewed

insight

on

a

rare

procedure

Miguel

Vieira

Marques

a,∗

,

João

Carneiro

a

,

Marta

Adriano

b

,

Filipa

Lanc

¸a

a

aServic¸odeAnestesiologia,HospitalUniversitáriodeSantaMaria,CentroHospitalardeLisboaNorte,E.P.E.,Lisboa,Portugal

bServic¸odeAnestesiologia,HospitalCurryCabral,CentroHospitalardeLisboaCentral,Lisboa,Portugal

Received29October2013;accepted4December2013 Availableonline10January2014

KEYWORDS

Airway---obstruction; Anesthesia---fetal; Anesthesia ---obstetric; Cervical lymphangioma; EXIT

Abstract Theexuterointrapartumtreatmentisararesurgicalprocedureperformedincases ofexpectedpostpartumfetalairwayobstruction.Thetechniqueliesonasafeestablishmentof apatentairwayduringlaborinanticipationofacriticalrespiratoryevent,withoutinterrupting maternal---fetalcirculation.

Anesthetic managementis substantially different from that regarding standard cesarean delivery anditsmaingoalsincludeuterine relaxation, fetal anesthesiaandpreservation of placentalbloodflow.

Wepresentthecaseofanexuterointrapartumtreatmentprocedureperformedonafetus withalargecervicallymphangiomaandprenatalevidenceofairwaycompromise.Modifications totheclassicexuterointrapartumtreatmentmanagementstrategiesweresuccessfullyadopted andwillbediscussedinthefollowingreport.

© 2013SociedadeBrasileirade Anestesiologia.Publishedby ElsevierEditoraLtda.Allrights reserved.

PALAVRAS-CHAVE

Viasaéreas ---obstruc¸ão; Anestesia---fetal; Anestesia ---obstétrica;

Linfangiomacervical; EXIT

Anestesiaparatratamentoex-úterointraparto:visãorenovadasobreum

procedimentoraro

Resumo Otratamentoex-úterointrapartoéumprocedimentocirúrgicofeitoemcasosraros deobstruc¸ãoesperadadasviasaéreasfetaisnopós-parto.Atécnicatemcomobaseo estab-elecimentosegurodeviasaéreaspermeáveisduranteotrabalhodepartoemantecipac¸ãoaum eventorespiratóriocrítico,seminterromperacirculac¸ãomaterno-fetal.

Omanejoanestésicoésubstancialmentediferentedaqueledestinadoàcesarianapadrãoe temcomoprincipaisobjetivosorelaxamentouterino,aanestesiafetaleapreservac¸ãodofluxo sanguíneoplacentário.

Apresentamoso caso deum procedimento para tratamentoex-útero intrapartofeito em fetocomumgrandelinfangiomacervicaleevidênciapré-nataldecomprometimentodasvias

Correspondingauthor.

E-mails:[email protected],[email protected](M.V.Marques).

(2)

526 M.V.Marquesetal.

aéreas.Asmodificac¸õesdasestratégiasadotadasnotratamentoex-úterointrapartoclássico foramfeitascomsucessoeserãodiscutidasnorelatoaseguir.

©2013SociedadeBrasileiradeAnestesiologia.PublicadoporElsevierEditoraLtda.Todosos direitosreservados.

Introduction

The ex utero intrapartum treatment (EXIT) is a rare sur-gicalprocedure performedtoensurefetalairwaypatency during labor in situations of expectedsevere, potentially life-threatening respiratory failure secondary to airway obstruction.

Also knownas operation on placental support1 (OOPS)

andairwaymanagementonplacentalsupport2(AMPS),EXIT

surgery was first described in the late 1980s by Norris

and colleagues3 and was initially performed in tracheal

occlusion reversion protocols for fetuses with congenital

diaphragmatichernia.4,5Theindisputableusefulnessofthis

technique later extended its applicability toa variety of

obstetricclinicalscenarios,6---8includingfetalheadandneck

tumorsurgicalapproaches.9

Theprocedureconsistsofapartialcesareansectionwith

simultaneousmaintenanceofplacentalcirculationasaway

topreserve fetal gas exchanges during the establishment

of a definitive airway through direct laryngoscopy,

bron-choscopy,ortracheostomy.

Theanestheticapproachissignificantlydifferentfroma

conventionalcesarean sectionandinvolvesadeepvolatile

anesthesiawithmaximumuterinerelaxation,preservation

ofuteroplacentalbloodflowandfetalanesthesia.

The success of an EXIT depends on a rigorous

strate-gicplanning withinvolvement of amultidisciplinary team

where the anesthesiologist often takes the leadership

role.

Inourreportwedescribetheanestheticmanagementofa

parturientscheduledforEXITsurgeryafterprenatal

diagno-sisofcervicallymphangiomawithmediastinalinvolvement,

highlightingbothfetalandmaternalsingularitiesinthelight

ofcurrentclinicalpractice.

Case

report

Ahealthy25-year-oldwoman,gravida1,para0,was

sched-uled for elective EXIT at 38 weeks of gestation due to

aprenatalultrasound diagnosis offetalcervical

lymphan-giomawithtrachealdeviationandriskofpostdeliveryairway

compromise.

Preparation for the procedure involved a

multidisci-plinaryteamof anesthesiologists,obstetricians,

neonatol-ogists,pediatricsurgeons,otolaryngologistsand

pulmonolo-gists. Several preliminary meetings were held and every

stakeholder’s role and positioning in the operatory room

wereclearlydefined.

Anesthesia material, room temperature, blood

group-ing,hemoderivativesavailabilityandbothneonatologyand

postanestheticcareunitvacancieswereallpreoperatively

confirmed.

Additional pharmacological preparation included

tocolyticsupportwithintravenousnitroglycerinsolutionat

aconcentrationof50mgmL−1anddrugsforsupplementary

intramuscularfetalanesthesia:fentanyl10␮gkg−1,

vecuro-nium0.2mgkg−1andatropine100

␮g,withatotalvolume

of2mL.

Standardmonitoringwasappliedwiththeparturientin

supine and leftlateral tilt positionunder manual uterine

displacement. Twointravenous 16Glineswere placedand

urinarycatheterizationwasperformed.

Balancedgeneralanesthesiawasinitiatedafter

preme-dicationwithfentanyl2␮gkg−1.Rapidsequenceinduction

was performed with propofol 2mgkg−1 and rocuronium

1.2mgkg−1, followed by endotracheal intubation and

mechanicalventilationinvolume-controlledmode.Aradial

arterycatheterwasplacedforinvasivebloodpressure

moni-toring.Anesthesiawasmaintainedwithlow-dosedesflurane

and nitrous oxide in oxygen mixture. Goal-directed fluid

therapywasmanagedwithcrystalloids.

Surgery began with a low segmental abdominal

inci-sionandhysterotomyfollowedbyfetalcephalicextraction

uptothe nippleline. Warm Hartmann’ssolution

amnioin-fusion was further initiated. The fetus’ airway was

exposed and evaluated by the neonatologist and

tra-cheal intubation successfully achieved after a single

attempt.

After full extraction the newborn was stabilized and

transportedinaneonatalincubatorundermechanical

ven-tilationtotheneonatologyunit.Totalplacentalbypasstime

was 4min and 46s. Amnioinfusion was discontinued and

uterinehypotonicityeffectivelyreversedwithoxytocinand

volatileconcentrationreduction.

The parturient remained hemodynamically stable

throughouttheprocedurewithMAP>70mmHg,equivalent

topreoperativerecords.Anesthesiaemergenceprogressed

uneventfully. Intravenous analgesia was performed with

paracetamol, ketorolac and tramadol and nausea and

vomitingprophylaxiswithdroperidol.

Discussion

Theidealconstitutionofamultidisciplinaryteam

interven-ingin an EXITsurgery isnotconsensual6,10,11 anddepends

onthe natureandpurposeof thesurgery:EXIT-to-airway,

EXIT-to-ECMOorEXIT-to-ressection.11

Inthiscase sixmedicalteams wereinvolved.

Anesthe-siology, obstetrics andneonatology weredirectly involved

intheprocedure. Additionalparticipationofpulmonology,

otolaryngology and pediatric surgery teams was justified

by their assistance in the event of a difficult

(3)

EXIT----renewedinsightonarareprocedure 527

establishasurgicalairwayorperformapartialtumor

resec-tion.

ThetwomainphysiologicalgoalsduringEXITareuterine

hipotonicitypreservation---whichfacilitatespartial

extrac-tionofthefetusandpreventsplacentaldissociation---and

placentalperfusionpressureconservationthatensuresfetal

oxygenation.1---15

Although associated with higher rates of morbidity

and mortality among global obstetric population,12

gen-eralanesthesiaisusuallyfavoredover regionaltechniques

in this procedure. Although not contraindicated, regional

anesthesia presents important disadvantages regarding

the cited precepts, particularly the risk of severe

hypotension and placental hypoperfusion.8 Aside from its

contribution to an adequate uterine relaxation, general

anesthesia allows simultaneous induction of both mother

and fetus through placental penetration of anesthetic

agents.

Maternal blood pressure must be keptwithin thelimit

of 10% of baseline.7,11 As a result, anesthesia induction

should be performed with the lowest possible

hemody-namicrepercussion,ideallyundercontinuousbloodpressure

monitoring.Inductionwithoutconsiderationforfetal

respi-ratorydepressionortimerestrictiontotheexpulsionhelps

to moderate the hypotensive effect of general

anesthet-ics.

Contrarytotheclassicrecommendationofdeep

inhala-tional anesthetic maintenance,6,13 the use of 0.5---1.0

halogenatedminimumanestheticconcentration(MAC),

sup-plementedifnecessarybyatocolytic,isbecomingaccepted

asaneffectivestrategyforuterinetonecontrol14with

min-imalcardiovascularimpactandlowerriskofpostoperative

uterineatony.6

Inthisparticularcase,pre-inductionopiate

administra-tion,rapidsequenceinduction withlowdosepropofoland

maintenancewith0.5MACdesfluraneprovidedanadequate

depthofanesthesia(BIS35---45)andastablehemodynamic

profile(MAP<10%).

The obtained uterine relaxation was in this case

con-sideredsufficientandintravenousnitroglycerin,previously

preparedasafirst-linetocolyticalternative,wasnot

admin-istered. Without uteroplacental circulation impairment,

fetalanesthesiawasenabledandadequatelydemonstrated

throughcompleteakinesiaatthetimeofairwayapproach,

alsoeliminatingtheneedfor supplementalexutero

anes-thesia.

After complete fetal extraction and umbilical cord

clamping, priorityfocused onpharmacologicalreversal of

uterinehipotonicitywithoxytocinandonfetusstabilization

forpediatricintensivecareunittransportation,whichwere

both held uneventfully. Cesarean section was completed

under close supervision of uterine contractility evolution

andhemostasis.

Developmentofahypocoagulablestatefollowinguterine

atony and massive postpartum bleeding has

occasion-ally been associated with the occurrence of epidural

hematoma.15 Considering the particular risks of

uter-ine atony and hemorrhage requiring blood transfusion

associated with EXIT surgery,7 we decided to replace

neuraxialregionalanalgesiafor aconventional

postopera-tiveintravenous analgesiaprotocol.Effective paincontrol

and patient satisfaction were observed. No reports of

hemorrhagicincidentswereissuedfollowingtheparturient’s

transferencetothepostanestheticcareunit.

Conclusion

The EXIT is an exceptional obstetric procedure bound for

life-savingfetalairwayinterventions.

PreparationforanEXITsurgeryinvolvesadetailed

multi-disciplinaryplanning,criticalfortheprocedure’ssafetyand

success.

Consideringcurrent literature,anesthesiamanagement

for this procedure is far from unequivocal. Even so and

despitetheadoptedstrategy,uteroplacentalcirculationand

uterusrelaxationaretobepreserveduntilthefetus’airway

issecured.

Anticipationofpostoperativecoagulationcomplications

mayprecludeuseofunrestrictedneuraxialanalgesic

tech-niques.

Authorship

Allauthorsparticipatedintheprocedurethatoriginatedthe

report.Datacollectionwasperformedbyall.Thereportwas

draftedbyM.M.andrevisedbyallauthors.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

1.SkarsgardED,ChitkaraU,KraneEJ,etal.TheOOPSprocedure (operationonplacentalsupport):inuteroairwaymanagement ofthefetuswithprenatallydiagnosedtrachealobstruction.J PediatrSurg.1996;31:826---8.

2.Collins DW, Downs CS, Katz SG, et al. Airway management on placental support (AMPS) --- the anaesthetic perspective. AnaesthIntensiveCare.2002;30:647---59.

3.Norris MC, Joseph J, Leighton BL. Anesthesia for perinatal surgery.AmJPerinatol.1989;6:39---40.

4.CrombleholmeTM,AlbaneseC.T.Thefetuswithairway obstruc-tion.Theunbornpatient.3rded.Philadelphia:Saunders,PA; 2001.p.357---71.

5.Flake AW, Crombleholme TM, Johnson MP, et al. Treatment of severe congenital diaphragmatic hernia byfetal tracheal occlusion:clinicalexperiencewithfifteencases.AmJObstet Gynecol.2000;183:1059---66.

6.Bouchard S, Johnson MP, Flake AW, et al. The EXIT proce-dure: experience and outcome in 31 cases. J Pediatr Surg. 2002;37:418---26.

7.TaghaviK,BeasleyS.Theexuterointrapartumtreatment(EXIT) procedure:applicationofanewtherapeuticparadigm.J Paedi-atrChildHealth.2013,http://dx.doi.org/10.1111/jpc.12223. 8.Choleva AJ.Anestheticmanagementofa patientundergoing

anexuterointrapartum treatment(EXIT) procedure:a case report.AANAJ.2011;79:497---503.

9.LiechtyKW, CrombleholmeTM,FlakeAW, etal. Intrapartum airwaymanagementforgiantfetalneckmasses:theEXIT(ex uterointrapartumtreatmentprocedure).AmJObstetGynecol. 1997;177:870---4.

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528 M.V.Marquesetal.

11.MarwanA,CrombleholmeT.M.TheEXITprocedure:principles, pitfalls,andprogress.SeminPediatrSurg.2006;15:107---15. 12.HawkinsJL, KooninLM,PalmerSK, etal. Anesthesia-related

deathsduringobstetricdeliveryintheUnitedStates1979---1990. Anesthesiology.1997;86:277---84.

13.Helfer DC,ClivattiJ,YamashitaAM,etal. Anesthesiafor ex uterointrapartumtreatment(EXITprocedure)infetuswith pre-nataldiagnosisoforalandcervicalmalformations:casereports. RevBrasAnestesiol.2012;62:411---23.

14.OkutomiT,SaitoM,KuczkowskiKM.Theuseofpotent inhala-tionalagents for theex-utero intrapartum treatment (EXIT) procedures: what concentrations? Acta Anaesthesiol Belg. 2007;58:97---9.

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