CASE
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InternationalJournalofSurgeryCaseReports62(2019)21–23ContentslistsavailableatScienceDirect
International
Journal
of
Surgery
Case
Reports
j o u r n al ho m e p a g e :w w w . c a s e r e p o r t s . c o mGiant
bulla
or
pneumothorax:
How
to
distinguish
Beatrice
Aramini
∗,
Ciro
Ruggiero,
Alessandro
Stefani,
Uliano
Morandi
DivisionofThoracicSurgery,DepartmentofMedicalandSurgicalSciencesforChildrenandAdults,UniversityofModenaandReggioEmilia,ViaLargodel Pozzon.71,41124,Modena,Italy
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Articlehistory: Received17June2019
Receivedinrevisedform26July2019 Accepted5August2019
Availableonline8August2019
Keywords: Pneumothorax Giantbulla Chestdrain Bullectomy
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BACKGROUND:Thedifferentialdiagnosis betweenpneumothoraxand giantbullaeisthoughttobe straightforwardbutsometimesposesachallenge.
CASEPRESENTATION:Wepresentacaseofa54-year-oldCaucasianmanwithagiantemphysematousbulla whounderwentsurgicalresection.Hehadnosmokinghistoryandhadpreviouspneumoniaepisodes. Thesurgerywasfreeofcomplications,withoutairleaks,andheshowedgoodventilationofthelung. DISCUSSION:Themaincomplicationsofbullaearepneumothorax,infectionandhemorrhage. Pneumoth-oraxisaseriouscomplicationinpatientswithcompromisedlungfunction.Therefore,itisveryimportant tocarefullydistinguishbullaefrompneumothoraxtoavoidiatrogenicpneumothoraxinpatientswith bullousdisease.
CONCLUSION:Weemphasizehowtodifferentiatebetweengiantbullaeandpneumothoraxutilizing history,physicalexamination,andradiologicalstudies,includingcomputedtomography(CT)scan.
©2019TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).
1. Background
Thedifferentiationbetweenpneumothorax anda giantbulla canbeverydifficultandoftenleadstoinaccuratediagnosisand management.Thiscasereportdemonstratestheclinicaland radi-ologicalpresentationofgiantbullaeandhighlightsthedifficulty makingadiagnosisandtreatingappropriately.Inparticular,we emphasizedhowtodifferentiatebetweengiantbullaeand pneu-mothoraxthroughhistory,physicalexamination,andradiological assessments,especiallycomputedtomography(CT)scan.Thiswork hasbeenreportedinlinewiththeSCAREcriteria[1].
2. Casepresentation
A54-year-oldmanwasreferredtotheoutpatientclinicofour Divisionforsurgicalresectionofgiantemphysematousbullae.He hadnosmokinghistory,andhehadpreviouspneumoniaepisodes andrecentlyexperiencedintermittentshortnessofbreath asso-ciatedwithpresyncopeepisodes.Sevendayspreviously,dyspnea worsened(MRCGradeIV)[2].Ongeneralinspection,thepatient wasalertandstable.Areviewofhismedicalrecordshoweda pro-gressivedeclineinlungfunctionwithforcedexpiratoryvolumein onesecond(FEV1)thatwas65%ofthevaluepredicted6months previouslyand FEV1 of 1.19Lthat was50% ofthat predicted6
∗ Correspondingauthor.
E-mailaddresses:beatrice.aramini@unimore.it
(B.Aramini),ciro.ruggiero@unimore.it(C.Ruggiero),alessandro.stefani@unimore.it
(A.Stefani),uliano.morandi@unimore.it(U.Morandi).
monthspreviously.ArterialbloodgasshowedapHof7.41, par-tialpressure ofCO2 (PCO2)of43mmHg,andpartialpressureof oxygen(PO2)of69mmHg.Thelefthemithoraxwashyperresonant topercussion,anddecreasedbreathsoundswereapparentupon auscultation.Thechestradiographyshowedprogressive enlarge-mentofthebullaintheleftlungofover10years(Fig.1A).ACT scanobtainedduringthecurrentadmissionrevealedagiantbulla withadiameterof10.5×11cmintheleftlobe,causingsignificant compressionoffairlynormallungparenchyma(Fig.1B).
Surgical criteria for resecting bullae are generally based on symptoms,particularlypoorpulmonaryfunction,inducing dysp-nea.Moreover,thegradeofdyspneawaspreviouslyclassified[3]: gradeIisminimaldyspneaonrunningoronexertingmorethan anordinaryeffort,gradeIIisdyspneaonordinaryeffort,gradeIIIis considerabledyspneaonexertinglessthananordinaryeffort,and gradeIVisdyspneaatrest.Inourcase,thepatientwasofgradeII. Thepatientunderwentsurgicalresectionofthebullafirstlyby VATSapproach,convertedafter30minintoaleftthoracotomyfor densepleuraladhesions(Fig.2AandB).Thebullapresentedasa sessileplant8–10cminlengthfromtheapicalsegmentoftheupper lobetothelowerlobeofthelung,anditappearedtobeofnormal consistency.Weproceededtodeflateandmobilizethebullafrom theparietalpleuraandmediastinaladhesions.Bullectomysection wasperformedbyGIAStapler-75andreinforcedwithinterrupted sutures ofVycril2/0(Fig.3A).Lysisofthepulmonary ligament wasperformed.Thepatientdidwellpostoperatively.Airleakage fromtheleftchesttubestoppedonthe3rdpostoperativeday,and thechesttubewasremoved.Thefollow-upchestX-rayshowed
https://doi.org/10.1016/j.ijscr.2019.08.003
2210-2612/©2019TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBYlicense(http://creativecommons. org/licenses/by/4.0/).
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22 B.Araminietal./InternationalJournalofSurgeryCaseReports62(2019)21–23
Fig.1. A.PreoperativechestX-ray;1BChest-CTshowingagiantbullaontheleftside,displacingthemediastinumontheright.Arrowsindicatethedouble-wallsigns.
Fig.2. A–B.DensepleuraladherencesvisibleonchestCT(arrows).
Fig.3. A.Surgicalisolationofthegiantbulla.Thebullameasured11cminlength.3BSix-daypostoperativechestX-ray.
nopneumothorax,andthepatientwasdischargedingoodgeneral clinicalconditiononthesixthday(Fig.3B).
3. Discussionandconclusion
A“bulla”isdefinedasanemphysematousspaceinthelungwith adiameterofmorethan1cminthedistendedstate[4].Giant bul-lousemphysema,originallydescribedbyBurke[5]in1937,isan idiopathic,distinctclinicalsyndromeofsevereprogressive dysp-neacausedbyextensive,predominantlyasymmetricupperlobe bullousemphysema,whichmayeventuallyleadtorespiratory fail-ure.Giantbullousemphysemahasalsobeencalledvanishinglung syndrome[5].Theradiographiccriteriaforgiantbullous emphy-sema,asdefinedbyRobertsetal.[6],includethepresenceofgiant bullaein oneorboth upperlobes, occupyingatleastone third ofthehemithoraxandcompressingthesurroundingnormallung
parenchyma.Sternetal.[7]describedtheCTfindingsofgiant bul-lousemphysema,whichincludemultiplelargebullae,rangingfrom 1to20cmindiameter(usually2–8cm),withoutasingledominant giantbulla.Radiologically,bullaeappearasavascularradiolucent areaswiththincurvilinearwalls.Thewallisusuallylessthan1mm inthicknessandmayevenbeinvisible,makingdetectionofthe bullaedifficult;theyaresometimesmistakenforpneumothorax. CTscansaremoresensitivethanchestx-raystodetectbullaefor theaccurateassessmentofthenumber,size,andpositionof bul-lae,especiallywhenthebullaeareobscured[7].Eligiblepatients includethosewithagiantbullaoccupyingone-fourthormoreof onehemithoraxonpreoperativeimaging.
Wedescribeinthisreportavaluablesigntodistinguish pneu-mothoraxfromadjacentgiantbullae:thedouble-wallsign.This signoccurswhen oneseesair outliningboth sidesof thebulla wallparalleltothechestwall(Fig.1AandB).Theabsenceofthis
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signprovidesfurtherevidenceand increasedconfidenceagainst thediagnosisofpneumothorax,which canpreventunnecessary chesttubeplacement.Thedouble-wallsignmaynotbeevidenton allCTslices,particularlywithcompressionofadjacentbullae,but carefulobservationofmultipleimageswillrevealthissignwhen apneumothoraxispresent.Onepotentialpitfallinthe apprecia-tionofthedouble-wallsignofpneumothoraxoccurswhentwo largebullaeareadjacenttooneanother(Fig.1B).Thissituationcan produceanapparentdouble-wallsign,mimickingpneumothorax. However,carefulscrutinyofmultipleimageswillshowtheabsence ofairinthepleuralspaceandthatthebullawallisnotparallelto thechestwallorparietalpleura.Themaincomplicationsofbullae arepneumothorax,infectionandhemorrhage.Pneumothoraxisa seriouscomplicationinpatientswithcompromisedlungfunction. Therefore,itisveryimportanttocarefullydistinguishbullaefrom pneumothoraxtoavoidiatrogenicpneumothoraxinpatientswith bullousdisease.
Funding
Nofunding.
Ethicalapproval
ForsinglecasereportNOethicalapprovalneeds.Patientsigned aconsentforpublishingthecasereport.
Consent
Patientsignedaconsentforthepublicationofthiscasereport.
Authorcontribution
BAandCRwrotethecasereport.ASandUMrevisedthecase report.
Registrationofresearchstudies
EthicalBoardapprovalisnotrequiredforcasereportsinour Center.
Guarantor
Prof.UlianoMorandiistheGuarantorofthiscasereport.
Provenanceandpeerreview
Notcommissioned,externallypeer-reviewed.
DeclarationofCompetingInterest
Theauthorshavenofinancialandpersonalrelationshipsto dis-close.
References
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[2]F.C.Chairman,Standardizedquestionnaireonrespiratorysymptoms:a statementpreparedandapprovedbyMRCCommitteeontheAetiologyof ChronicBronchitis,BMJ2(1665)1960.
[3]TheCriteriaCommitteeoftheNewYorkHeartAssociation,Nomenclatureand CriteriaforDiagnosisofDiseasesoftheHeartandGreatVessels,9thed.,Little, Brown&Co,Boston,1994,pp.253–256.
[4]M.N.Zahara,etal.,Onemaydiefromgiantbullae,HeartViews8(June–August (2))(2005)62–65.
[5]R.Burke,Vanishinglungs:acasereportofbullousemphysema,Radiology28 (367)(1937)-371.
[6]L.Roberts,C.E.Putman,J.T.T.Chen,L.R.Goodman,C.E.Ravin,Vanishinglung syndrome:upperlobebullouspneumopathy,Radiol.Interam.Radiol.12 (1987)249–255.
[7]E.J.Stern,W.R.Webb,A.Weinacker,N.L.Muller,Idiopathicgiantbullous emphysema(vanishinglungsyndrome):imagingfindingsinninepatients,AJR 162(1994)279–282.
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