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CASEREPORTOpenAccessHybridvideo-assistedthoracoscopicsurgerylobectomyoffissurelesscongenitalcysticadenomatoidmalformation:acasereportMitsuyukiNakata1*,ShigetoshiYoshida2,TakeshiSaito1,KeitaTerui1,TetsuyaMitsunaga1,SachieOhno1,NaokoMise1,SatoruOita1andHideoYoshida1AbstractIntroduction:Thoracoscopiclobectomyforcongenitalpulmonaryairwaymalformationhasbeenindicatedfromtheneonatalperiodtoadolescence.
However,itisdifficulttoapproachthepulmonaryarteryforlobectomyincongenitallungmalformationswithincompleteorabsentinterlobarfissures.
Multidetectorcomputedtomographicimagesandcomputedtomographypulmonaryangiographygaveushelpfulinformationbeforetheoperation.
Weperformedthoracoscopiclobectomyforcongenitalpulmonaryairwaymalformationswithabsentinterlobarfissuresandadhesionsinaccordancewithinformationfrommultidetectorcomputedtomographicimages.
Casepresentation:A14-year-oldJapanesegirlreceivedadiagnosisofcongenitalpulmonaryairwaymalformationwhenshepresentedwithpneumonia.
Usingmultidetectorcomputedtomographyandthree-dimensionalreconstructionprovidesmeticulouscharacterizationoftheanatomyinpediatricpatients.
WeconfirmedthatherleftA4+5arteryarosefromherleftpulmonaryarterymedialtoA6.
HerleftpulmonaryarterywasdividedjustproximaltotheA6originbeforethelobeswereseparatedsafely.
Wetookadvantageofusingastaplertodividethefissurelessthickparenchyma.
Perioperativediagnosiswascongenitalcysticadenomatoidmalformation.
Conclusions:Weusedpreoperativemultidetectorcomputedtomographytooutlinethebronchovascularanatomyandguidehybridvideo-assistedthoracoscopicsurgeryforacongenitalcysticadenomatoidmalformationinafissurelessleftlung.
Keywords:Congenitalcysticadenomatoidmalformationoflung,HybridVATS,ThoracicsurgeryIntroductionCongenitalpulmonaryairwaymalformation(CPAM)hasbeenhistoricallydiagnosedinabroadrangeofagegroups,fromtheprenatalperiodtoadulthood.
ThespectrumofCPAMrequiringsurgeryincludescongenitalcysticadeno-matoidmalformation(CCAM),intra-andextrapulmonarysequestration,andsymptomaticcongenitallobaremphy-sema.
TheindicationsforsurgeryofasymptomaticCPAMarecontroversial[1].
Theriskofinfectioninthefirst3monthsafterbirthislow.
Stantonetal.
reportedthatasymptomaticantenatallydiagnosedinfantsdevelopedsymptomsatamedianageof6.
9months(range,2to10months)[2].
Pelizzoetal.
reportedchronicinflammationin50%ofasymptomaticCPAMsresectedat3monthsofage[3].
Thissuggeststhatlobectomyinneonatesorinfantsisdesirable,eveninasymptomaticcases.
Althoughtherearestillfewreportsofthoracoscopiclobectomyinchildren,thoracoscopiclobectomyforCPAMhasbeenindicatedfromtheneonatalperiodtoadolescence[4-6].
However,adhesionsandincompleteorabsentinterlobarfissuresmakethoracoscopicsurgerydifficult,eveninadults[7].
Inthispaper,wedescribeusingmultidetectorcomputedtomography(MDCT)three-dimensionalreconstructionstoguideahybridvideo-assistedthoracoscopicsurgery(VATS)lobectomysafelyinapatientwithabsenceoftheinterlobarfissure,andprovideabriefreviewoftheexistingliterature.
*Correspondence:mitchinakachi@gmail.
com1DepartmentofPediatricSurgery,GraduateSchoolofMedicine,ChibaUniversity,1-8-1Inohana,Chuo-ku,Chiba260-8677,JapanFulllistofauthorinformationisavailableattheendofthearticleJOURNALOFMEDICALCASEREPORTS2015Nakataetal.
;licenseeBioMedCentral.
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0),whichpermitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycredited.
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Nakataetal.
JournalofMedicalCaseReports2015,9:23http://www.
jmedicalcasereports.
com/content/9/1/23CasepresentationA14-year-oldJapanesegirlreceivedadiagnosisofCPAMwhenshepresentedwithpneumonia.
Volumet-ricthin-sectionhigh-resolutioncomputedtomography(CT)revealedalmostcompleteabsenceoftheleftinter-lobarfissure(Figure1).
Alesionwasdetectedinthebasalsegmentofherleftlowerlobe,consistingofmul-tiplecystsbutnoabnormalvessels.
ThepreoperativediagnosiswasCCAM.
CTpulmonaryangiographywiththree-dimensionalreconstructionsofherpulmonaryvesselswasperformed.
Imageswereacquiredusinga16-slicescanner(Light-SpeedUltra;GEHealthcare,MilwaukeeWI,USA).
Iohexol(400mgI/kgofcontrastmedium,toamaximumof100mL)wasinjectedintravenouslyatarateof2mL/second.
Imagesrevealedherleftlingualarterytobethefirstbranchofherleftmainpulmonaryartery,whichistheso-calledmediastinallingualartery(Figure2).
HybridVATSlobectomyusingdifferentiallungventila-tionwasperformedintherightlateraldecubitusposition.
Weusedasmallthoracotomyincision(5.
5cm)alongthefifthintercostalspacecenteredonthemidaxillaryline,andtwoportsinthesixthintercostalspacealongthepost-axillarylineandintheseventhintercostalspacealongthemidaxillaryline.
AsseenonCT,herleftlunghadalmostnofissure.
Herleftinferiorpulmonaryveinwasfirstdividedusingastapler(Endo-GIAwhite,Covidien,MansfieldMA,USA).
Itwasdifficulttofreetheaffectedarteriesandbronchifromextensiveadhesions.
Afterwelocatedtheleftupperandlowerbronchiusingabroncho-scope,weconfirmedtheCTfindingofthemediastinallingualarteryarisingmedialtoA6.
Beforethefissureswereseparated,theleftlowerbronchuswasdividedusingastapler(Endo-GIApurple)andherleftpulmonaryarterywasdivided,alsowithastapler(Endo-GIAwhite),justproximaltotheoriginofA6.
Afterherleftlungwasinflatedbyananesthesiologist,theborderlinebetweenthedeflatedandtheinflatedareaclearlyappeared(Figure3).
Herleftlowerlungwasdividedwithstaplers(Endo-GIApurpleonetimeandblackfourtimes).
Theprocedurelasted5hours22minuteswithasmallamountofbloodloss.
Therewerenocomplications.
DiscussionThoracoscopiclobectomyforCPAMsinpediatricpatientsispracticalandsafe.
Patientsaregenerallypleasedwiththecosmeticresult.
Thegoodviewofthehilarareaensureslessbloodloss.
NasrandBassreportedthattherewasnosignificantdifferencebetweenthoracoscopicandopenproceduresintherateofoverallcomplicationsandthedurationofsurgerybymeta-analysis[8].
OuroperationFigure1High-resolutioncomputedtomographyofthelung.
Congenitalpulmonaryairwaymalformationwasdetectedinthebasalsegmentofleftinferiorlobewithaseverelyatreticinterlobarfissure.
Thelesionconsistedofmultiplecystswithnoabnormalvessels.
Figure2Three-dimensionalcomputedtomographyofthepulmonaryartery,tracheaandbronchus.
Computedtomographicpulmonaryangiographywiththree-dimensionalreconstructionsshowingtheleftA4+5artery(themediastinallingualartery)arisingmedialtoA6.
Redimage,pulmonaryartery;yellowimage,tracheaandbronchus;whitearrow,A4+5(themediastinallingualartery);whitearrowhead,A6;asterisk,commonbasalartery.
Figure3Operativeprocedure.
Almostnofissureintheleftlungwasidentifiableafterthearteries,veins,andbronchiweredivided.
Afterisolatinganddividingtheleftinferiorpulmonaryvein(A),thelowerbronchus(B)andthepulmonaryartery(C:arrowhead,A6;asterisk,commonbasalartery),theresiduallungwasinflatedtovisualizetheborderwiththelowerlobe(D:whitedottedline).
Nakataetal.
JournalofMedicalCaseReports2015,9:23Page2of4http://www.
jmedicalcasereports.
com/content/9/1/23washamperedbyadhesions(probablyduetopreviousinfections)[8]andanatreticinterlobarfissure.
Inthecaseofincompleteinterlobarfissuresorfusedfissuresitisdifficulttodeterminethearterialanatomy,andforcibledivisionoffusedfissuresrisksvesselinjuryandprolongedairleakage[9].
WeselectedhybridVATS.
Aminithoracotomycom-binedwithvideoassistperformedpredominantlyviadirectvisualizationwasasecure,integrated,minimallyinvasiveapproachtothelargefusedfissureandthesevereadhesion[10].
Preoperativeimagingstudiesareessentialforassessingnotonlythecongenitallungdiseasebutalsotheanat-omyofvessels,bronchi,andfissures.
UsingMDCTandthree-dimensionalreconstructionprovidesmeticulouscharacterizationoftheanatomyinpediatricpatients.
AccordingtoLeeetal.
,typesandlocationofcongenitallungdiseaseandanomalousvesselsassociatedwithcon-genitallunganomaliesweredetectedwithhighaccuracy[11].
Inourcase,weknewthattheleftA4+5arterywasthemediastinallingualarteryandtheleftpulmonaryarterywasdividedjustproximaltotheA6originbeforethefissureswereseparated.
Duringtheoperationitmaybenecessarytoproduceafissuralseparation.
Inadvertentseparationresultsinpro-longedairleakage.
Ithasbeenreportedthattheuseofvariousdevices,suchasstaplers,bipolarthermofusion,biodegradablesealant,andultrasonicallyactivatedscal-pels[12]canpreventprolongedairleakage.
Automaticstaplingdeviceshavebeenoftenusedforinterlobarfissuredivisionforpulmonarylobectomy.
Thomasetal.
explainedhowtousestaplerssafelytodividefissures[13].
Usingstaplersishelpfulindividingthickparen-chymawithnofissure.
Nomorietal.
reportedthattherewasnopostoperativeairleakageinthepatientsafterlobectomywithlargefusedfissuresdividedbystaplers.
Theydefinedthestepsoftheproceduresaccordingtoeachlobectomy[7].
Thomasetal.
introducedthetech-niqueofdividingtheparenchymawithstaplerstoexposethepulmonaryarteries[13].
Inneonatesandinfantstheoperativefieldistoosmalltouseastapler.
Albaneseetal.
recommendedbipolarelectrocoagulation(LigaSureCovidien,MansfieldMA,USA)forsealingpulmonaryvesselsanddividingfissuresduringlobectomyininfants.
Intheirseries,theyusedthisonbotharteriesandveinslessthan7mmindiameterbecauseofthelowpressurepulmonarycirculationinsmallchildren[14].
Santinietal.
assessedtheefficacyandsafetyofbipolarelectrocoagulationinexperimentalstud-ieswithanimalsandhumans;theyfounditwasusefulindividingfissures[15].
Kanekoetal.
suggestedthatbipolarelectrocoagulationseemedtohaveaweakersealingeffectunderwetconditions,andtherewasanincreasedriskofhemorrhage[16].
ConclusionsInsummary,wereportedhybridvideo-assistedthoraco-scopicsurgicallobectomyoffissurelessCCAMfora14-year-oldgirl.
ThoracoscopiclobectomyforCPAMhasbeenindicatedfromtheneonatalperiodtoadolescence.
However,itisdifficulttoapproachthepulmonaryarteryforlobectomyincongenitallungmalformationswithincompleteorabsentinterlobarfissures.
MDCTandCTpulmonaryangiographygaveushelpfulinformationbe-foretheoperation.
WeconfirmedthathermediastinallingualarteryarosemedialtoA6.
HerleftpulmonaryarterywasdividedjustproximaltotheA6originbeforethelobeswereseparatedsafely.
Wetookadvantageofusingastaplertodividethefissurelessthickparen-chyma.
WeperformedsafelyhybridVATSlobectomyonanessentiallyfissurelesslungusingstaplers,guidedbypreoperativeMDCTandCTpulmonaryangiography.
ConsentWritteninformedconsentwasobtainedfromthepatient'sparentforpublicationofthiscasereportandaccompany-ingimages.
AcopyofthewrittenconsentisavailableforreviewbytheEditor-in-Chiefofthisjournal.
CompetinginterestsTheauthorsdeclarethattheyhavenocompetinginterests.
Authors'contributionsAllauthorscontributedtorevisionofthereport.
Allauthorsreadandapprovedthefinalmanuscript.
AcknowledgementsTheauthorshavenodisclosurestomakeandhavehadnosourceoffundinginthepreparationofthismanuscript.
WewouldliketothankDrShigetoshiYoshidaforhistechnicalsupport.
Authordetails1DepartmentofPediatricSurgery,GraduateSchoolofMedicine,ChibaUniversity,1-8-1Inohana,Chuo-ku,Chiba260-8677,Japan.
2DepartmentofGeneralThoracicSurgery,GraduateSchoolofMedicine,ChibaUniversity,1-8-1Inohana,Chuo-ku,Chiba260-8677,Japan.
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