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SpontaneousRuptureofaMediastinalBronchialArteryAneurysmInducedbyAnticoagulantAgentZhiminWang1ChenghuaXu1XiaoxiaoDing2JinyingChen1HuapingXin11TheFirstPeople'sHospitalofTaizhou,Taizhou,Zhejiang,China2TheCentreHospitalofTaizhou,Taizhou,Zhejiang,ChinaThoracCardiovascSurgRep2016;5:18–20.
AddressforcorrespondenceHuapingXin,MD,TheFirstPeople'sHospitalofTaizhou,No.
218,HengjieRoad,TaizhouNo.
218,Hengji,China(e-mail:xhp691@163.
com).
JinyingChen,PD,TheFirstPeople'sHospitalofTaizhou,No.
218,HengjieRoad,TaizhouNo.
218,Hengji,China(e-mail:498024616@qq.
com).
IntroductionAcutehemomediastinumisusuallycausedbyruptureofachesttrauma,aortaandvertebralarterydissection,andatumor.
1Itcanalsooccurasaresultofsurgery,angiographyandsomedrugtreatments.
2,3However,nontraumaticspon-taneousruptureofbronchialarteryaneurysm(BAA)4isveryrare.
TherearefewreportsaboutthespontaneousruptureofaBAAcausedbyusinganticoagulantagentofpatients,whohavenothadanymedicalhistoryoftrauma,hypertension,orapparentaorticpathology.
Inthisreport,wearegoingtodiscussacaseofanacutehemomediasti-numcausedbyarupturedBAA.
Thepatientinthecasewasusinglow-molecular-weightheparinandwarfarintotreatdeepveinthrombosis(DVT).
Toourknowledge,thesituationinthecasehasnotbeenreportedinanyliteratureyet.
CaseReportA66-year-oldmanwhohasamedicalhistoryofpulmonaryheartdiseasewasadmittedtohospitalbecauseofprogressiveshortnessofbreathandleftlowerextremityedemaover3days.
Hisinitialvitalsignswereasfollows:bloodpressure(BP),146/105mmHg;pulse,94beats/min;respiration,20breaths/min;andtemperature,36.
5°C.
Hedeniedhehadbeenatobaccouser.
Hedidnothaveafeveronadmission.
Therewerenosignsorsymptomsofradiatingbackpain,chestpain,orthopnea,hoarseness,ordysphagia.
Laboratorytestresultsindicatednothingabnormal(andtheinternationalnormalizedratio[INR]was1.
08).
Acomputedtomography(CT)scanofthechestshowedthatbothsidesofbronchiahadsignsofinfection.
Inaddition,avenousultrasoundshowedtherewasDVTonhisleftlowerextremity.
Therefore,thepatientwastreatedforvenousthrombosisinhospitalwithKeywordsbronchialarteryaneurysmanticoagulantagentspontaneousruptureAbstractNontraumaticspontaneousruptureofabronchialarteryaneurysmisrarelyseen.
Inthisreport,wedescribedsuchaphenomenoninapatientinducedbyusageofanticoagulantagent.
Thepatienthadnoantecedenthistoryoftrauma,hypertension,orapparentaorticpathology.
Thepatientwhohadbeentakinglow-molecular-weightheparinandwarfarintotreatdeepveinthrombosiscomplainedofasuddenupperabdomenpainwithshortnessofbreathandhypoxemia.
Thepatientwasdiagnosedandtreatedforanacutehemomediastinumcausedbyarupturedbronchialarteryaneurysm.
Ifthepatienthadcontinuedtotaketheanticoagulantantithromboticdrugs,itmaycauseamorevirulentbleeding.
Takentogether,CTangiographyisausefuldiagnosistoolforpatientswithsuddenchestpainandabdominalpain,andrarecauseshouldbeconsidered.
receivedDecember7,2015acceptedafterrevisionJanuary13,2016publishedonlineApril4,2016DOIhttp://dx.
doi.
org/10.
1055/s-0036-1578813.
ISSN2194-7635.
2016GeorgThiemeVerlagKGStuttgart·NewYorkCaseReport:ThoracicTHIEME18low-molecular-weightheparin(4,100U/dbyhypodermicinjectionfor3days)andwarfarin(3mg/dorallyforalongerperiodoftime).
Withthistreatment,thepatientshowedsomeimprovementsofhissymptoms:nomoreshortnessofbreathandreducedlowerextremityedema.
Onthe10thdayofthistreatment,thepatientcomplainedofsuddenupperabdomenpain.
Healsostartedtovomitandcoldsweating.
Afterhewasassessed,hewasafebrile.
BPwas83/44mmHg,andheartratewas89beats/min.
Electrocardiogramindicatedcompletedrightbundlebranchblock.
Arterialbloodgasanalysisshowedthaton100%oxygen,potentield'hydrogene(pH)was7.
34,partialpressureofoxygen(PO2)was77mmHg,andpartialpressureofcarbondioxide(PaCO2)was55.
1mmHg.
Thepatienthadobviousstridor,andhischestwasbilateralwheezing.
Laboratorytestresultsindicat-ednothingabnormal,exceptforanelevatedD-dimerlevelof2.
05μg/mL(referencerange,0–0.
5μg/mL)andINRof2.
05.
CTscanofthechestshowedmediastinalbleeding(Fig.
1B).
CTofthepulmonaryarteryrevealedmediastinalhematoma(Fig.
1D–F).
AorticarteryCTdemonstratedaruptureontherightbronchusartery.
Atlast,aselectivearteriography(Fig.
1G)furtherconrmedthattherewasanacutebilateralhemothoraxsecondarytoarupturedBAAoftherightbronchialartery.
Thebleedingwasstoppedafterdiscontinuationoflow-molecular-weightheparinandwarfarin.
Therefore,aruptureoftherightbronchialarterywithmediastinalhematomaformationwasdiagnosed.
Thenthebronchusarteryaneurysmwassuccessfullytreatedbycoilembolization(Fig.
1H).
Finally,thepatientwasturningbetter.
DiscussionInmostcasesofnontraumaticmediastinalhemorrhage,therearesignsofchestpainordyspnea.
However,hemorrhagicshockisrare.
1Moreover,arupturedBAAisarareetiologyformediastinalhemorrhage.
1BAAisarareentitythatisobservedinlessthan1%ofallcasesofselectivebronchialarteriography.
5Interestingly,ourpatienthadasuddenupperabdomenpain,hypoxemia,andshockaftertakingananticoagulantagent.
Inthiscase,theinitialmisdiagnosiswasmainlyduetotheclinicalpresentationsofasuddenupperabdomenpain,worsenedshortnessofbreath,hypoxemia,andDVToftheleftlowerextremity,whicharealsothemostcommonsymptomsandriskfactorsamongpatientswithinitialpulmonaryembolism.
Wesystematicallyreviewedtheliteraturefor"hemome-diastinum"and"rupturedBAA"onPubMed.
SincetherstcasereportedbyPugnalein2001,6onlynineadditionalcaseshavebeenreporteduntil2014.
7InthepathogenesisofBAAs,chronicinammationofthelungisanimportantcontribut-ingfactor(tuberculosis,histoplasmosis,aspergillosis,bron-chiectasis,etc.
).
8,9Inourcase,thecausewaslikelythechronicinammationofbronchiectasis.
Unfortunately,anticoagulantagentcaninducespontaneousruptureoftherightBAA,andthenresultsinhemomediastinum.
BAAsaretypicallyclassiedanatomically,eitherasmediasti-nalorintrapulmonary,accordingtotheirdifferentassociatedclinicalsymptoms.
Intrapulmonaryaneurysmsoftenpresentwithmassiveorintermittenthemoptysis.
Althoughmediastinalaneurysmsmaymanifestasamediastinalmass,acutesuperiorvenacavaobstruction,dysphagia,hemothorax,hemomediasti-num,andhematemesiscanoccur.
10Onimaging,hemothoraxandhemomediastinumarethemostcommonndings1;CTangiographyistheprimarynoninvasivediagnosticmodalityforBAAs.
11However,conventionalangiographycanbeperformedforbothdiagnosticandtreatmentpurposes.
5Nowadays,endo-vasculartechniquessuchastranscatheterembolizationhasbeenincreasinglyapplied.
Insummary,herewereportedararecaseofrupturedBAA,whichhasnotbeendescribedbefore.
CorrectdiagnosisofthisproblemcanbeobtainedbyCTangiographyandselectivearteriography.
ThispatienthadahistoryofDVT.
Therefore,whenhesuddenlyhadabdominalpain,wespontaneouslyassumedthathehadpulmonaryembolism,whileignoringothercauses.
Ifthepatienthadcontinuallytakenanticoagu-lantantithromboticdrugs,avirulentbleedingwouldhaveFig.
1(A)TherewasnomediastinalbleedingintheCTscanofthechestwhenthepatientwasadmitted.
(B)TherewasamediastinalbleedingintheCTscanafterthepatientexperiencedsuddenupperabdomenpain.
(D–F)AorticarteryCTdemonstratedaruptureontherightbronchusartery.
(G)Aselectivearteriographyfurtherconrmedthattherewasanacutebilateralhemothoraxsecondarytoarupturedbronchialarteryaneurysmoftherightbronchialartery.
(H)Thebronchusarteryaneurysmwassuccessfullytreatedbycoilembolization.
ThoracicandCardiovascularSurgeonReportsVol.
5No.
1/2016AnticoagulantAgent–InducedRuptureofaMediastinalBAAWangetal.
19occurred.
Takentogether,commonpresentationischestpain,whichmayleadtoconfusionwithothersyndromes,suchaschestwalldiseasesanddigestivesystemdiseases,soCTangiographyisnecessaryfordifferentialdiagnosisofchestpainandabdominalpain.
ConictofInterestTheauthorsdeclarethatthereisnoconictofinterest.
FundingThisworkwasnotsupportedbyanyfunding.
Reference1SeoYH,KwakJY.
Spontaneoushemomediastinumandhemo-thoraxcausedbyarupturedbronchialarteryaneurysm.
KoreanJThoracCardiovascSurg2011;44(4):314–3172TailléC,FartoukhM,HoulR,KobeiterH,RémyP,LemaireF.
Spontaneoushemomediastinumcomplicatingsteroid-inducedmediastinallipomatosis.
Chest2001;120(1):311–3133YokoyamaH,OhmiM,SadahiroM,ShojiY,TabayashiK,MoizumiY.
Spontaneousruptureofthethoracicaorta.
AnnThoracSurg2000;70(2):683–6894MatsugeS,HosokawaY,MurakamiY,SatohK.
[Thymomawithidiopathicthrombocytopenicpurpuraprecededbymediastinalhemorrhage;reportofacase].
KyobuGeka2002;55(10):899–9025MizuguchiS,InoueK,KidaA,etal.
Rupturedbronchialarteryaneurysmassociatedwithbronchiectasis:acasereport.
AnnThoracCardiovascSurg2009;15(2):115–1186PugnaleM,PortierF,LamarreA,etal.
Hemomediastinumcausedbyruptureofabronchialarteryaneurysm:successfultreatmentbyembolizationwithN-butyl-2-cyanoacrylate.
JVascIntervRadiol2001;12(11):1351–13527VosseBA,vanBelleAF,deVriesGJ,DasM.
Hemomediastinumduetospontaneousruptureofamediastinalbronchialarteryaneu-rysm—ararecauseofthoracicpain.
RespirMedCaseRep2014;12:27–298ShihSY,HuSY,TsanYT,LinTC.
Spontaneoushemomediastinumandhemothoraxcausedbyrupturedbronchialarteryaneurysm.
EurJCardiothoracSurg2009;35(6):10769ChatterjeeA,GhoshS,SalhiyyahK,GainesP,RoccoG.
Ararepresentationofarupturedbronchialarteryaneurysm.
Thorax2004;59(10):91210WilsonSR,WingerDI,KatzDS.
CTvisualizationofmediastinalbronchialarteryaneurysm.
AJRAmJRoentgenol2006;187(5):W544-511HoffmannV,YsebaertD,DeSchepperA,ColpaertC,JorensP.
Acutesuperiorvenacavaobstructionafterruptureofabronchialarteryaneurysm.
Chest1996;110(5):1356–1358ThoracicandCardiovascularSurgeonReportsVol.
5No.
1/2016AnticoagulantAgent–InducedRuptureofaMediastinalBAAWangetal.
20
AddressforcorrespondenceHuapingXin,MD,TheFirstPeople'sHospitalofTaizhou,No.
218,HengjieRoad,TaizhouNo.
218,Hengji,China(e-mail:xhp691@163.
com).
JinyingChen,PD,TheFirstPeople'sHospitalofTaizhou,No.
218,HengjieRoad,TaizhouNo.
218,Hengji,China(e-mail:498024616@qq.
com).
IntroductionAcutehemomediastinumisusuallycausedbyruptureofachesttrauma,aortaandvertebralarterydissection,andatumor.
1Itcanalsooccurasaresultofsurgery,angiographyandsomedrugtreatments.
2,3However,nontraumaticspon-taneousruptureofbronchialarteryaneurysm(BAA)4isveryrare.
TherearefewreportsaboutthespontaneousruptureofaBAAcausedbyusinganticoagulantagentofpatients,whohavenothadanymedicalhistoryoftrauma,hypertension,orapparentaorticpathology.
Inthisreport,wearegoingtodiscussacaseofanacutehemomediasti-numcausedbyarupturedBAA.
Thepatientinthecasewasusinglow-molecular-weightheparinandwarfarintotreatdeepveinthrombosis(DVT).
Toourknowledge,thesituationinthecasehasnotbeenreportedinanyliteratureyet.
CaseReportA66-year-oldmanwhohasamedicalhistoryofpulmonaryheartdiseasewasadmittedtohospitalbecauseofprogressiveshortnessofbreathandleftlowerextremityedemaover3days.
Hisinitialvitalsignswereasfollows:bloodpressure(BP),146/105mmHg;pulse,94beats/min;respiration,20breaths/min;andtemperature,36.
5°C.
Hedeniedhehadbeenatobaccouser.
Hedidnothaveafeveronadmission.
Therewerenosignsorsymptomsofradiatingbackpain,chestpain,orthopnea,hoarseness,ordysphagia.
Laboratorytestresultsindicatednothingabnormal(andtheinternationalnormalizedratio[INR]was1.
08).
Acomputedtomography(CT)scanofthechestshowedthatbothsidesofbronchiahadsignsofinfection.
Inaddition,avenousultrasoundshowedtherewasDVTonhisleftlowerextremity.
Therefore,thepatientwastreatedforvenousthrombosisinhospitalwithKeywordsbronchialarteryaneurysmanticoagulantagentspontaneousruptureAbstractNontraumaticspontaneousruptureofabronchialarteryaneurysmisrarelyseen.
Inthisreport,wedescribedsuchaphenomenoninapatientinducedbyusageofanticoagulantagent.
Thepatienthadnoantecedenthistoryoftrauma,hypertension,orapparentaorticpathology.
Thepatientwhohadbeentakinglow-molecular-weightheparinandwarfarintotreatdeepveinthrombosiscomplainedofasuddenupperabdomenpainwithshortnessofbreathandhypoxemia.
Thepatientwasdiagnosedandtreatedforanacutehemomediastinumcausedbyarupturedbronchialarteryaneurysm.
Ifthepatienthadcontinuedtotaketheanticoagulantantithromboticdrugs,itmaycauseamorevirulentbleeding.
Takentogether,CTangiographyisausefuldiagnosistoolforpatientswithsuddenchestpainandabdominalpain,andrarecauseshouldbeconsidered.
receivedDecember7,2015acceptedafterrevisionJanuary13,2016publishedonlineApril4,2016DOIhttp://dx.
doi.
org/10.
1055/s-0036-1578813.
ISSN2194-7635.
2016GeorgThiemeVerlagKGStuttgart·NewYorkCaseReport:ThoracicTHIEME18low-molecular-weightheparin(4,100U/dbyhypodermicinjectionfor3days)andwarfarin(3mg/dorallyforalongerperiodoftime).
Withthistreatment,thepatientshowedsomeimprovementsofhissymptoms:nomoreshortnessofbreathandreducedlowerextremityedema.
Onthe10thdayofthistreatment,thepatientcomplainedofsuddenupperabdomenpain.
Healsostartedtovomitandcoldsweating.
Afterhewasassessed,hewasafebrile.
BPwas83/44mmHg,andheartratewas89beats/min.
Electrocardiogramindicatedcompletedrightbundlebranchblock.
Arterialbloodgasanalysisshowedthaton100%oxygen,potentield'hydrogene(pH)was7.
34,partialpressureofoxygen(PO2)was77mmHg,andpartialpressureofcarbondioxide(PaCO2)was55.
1mmHg.
Thepatienthadobviousstridor,andhischestwasbilateralwheezing.
Laboratorytestresultsindicat-ednothingabnormal,exceptforanelevatedD-dimerlevelof2.
05μg/mL(referencerange,0–0.
5μg/mL)andINRof2.
05.
CTscanofthechestshowedmediastinalbleeding(Fig.
1B).
CTofthepulmonaryarteryrevealedmediastinalhematoma(Fig.
1D–F).
AorticarteryCTdemonstratedaruptureontherightbronchusartery.
Atlast,aselectivearteriography(Fig.
1G)furtherconrmedthattherewasanacutebilateralhemothoraxsecondarytoarupturedBAAoftherightbronchialartery.
Thebleedingwasstoppedafterdiscontinuationoflow-molecular-weightheparinandwarfarin.
Therefore,aruptureoftherightbronchialarterywithmediastinalhematomaformationwasdiagnosed.
Thenthebronchusarteryaneurysmwassuccessfullytreatedbycoilembolization(Fig.
1H).
Finally,thepatientwasturningbetter.
DiscussionInmostcasesofnontraumaticmediastinalhemorrhage,therearesignsofchestpainordyspnea.
However,hemorrhagicshockisrare.
1Moreover,arupturedBAAisarareetiologyformediastinalhemorrhage.
1BAAisarareentitythatisobservedinlessthan1%ofallcasesofselectivebronchialarteriography.
5Interestingly,ourpatienthadasuddenupperabdomenpain,hypoxemia,andshockaftertakingananticoagulantagent.
Inthiscase,theinitialmisdiagnosiswasmainlyduetotheclinicalpresentationsofasuddenupperabdomenpain,worsenedshortnessofbreath,hypoxemia,andDVToftheleftlowerextremity,whicharealsothemostcommonsymptomsandriskfactorsamongpatientswithinitialpulmonaryembolism.
Wesystematicallyreviewedtheliteraturefor"hemome-diastinum"and"rupturedBAA"onPubMed.
SincetherstcasereportedbyPugnalein2001,6onlynineadditionalcaseshavebeenreporteduntil2014.
7InthepathogenesisofBAAs,chronicinammationofthelungisanimportantcontribut-ingfactor(tuberculosis,histoplasmosis,aspergillosis,bron-chiectasis,etc.
).
8,9Inourcase,thecausewaslikelythechronicinammationofbronchiectasis.
Unfortunately,anticoagulantagentcaninducespontaneousruptureoftherightBAA,andthenresultsinhemomediastinum.
BAAsaretypicallyclassiedanatomically,eitherasmediasti-nalorintrapulmonary,accordingtotheirdifferentassociatedclinicalsymptoms.
Intrapulmonaryaneurysmsoftenpresentwithmassiveorintermittenthemoptysis.
Althoughmediastinalaneurysmsmaymanifestasamediastinalmass,acutesuperiorvenacavaobstruction,dysphagia,hemothorax,hemomediasti-num,andhematemesiscanoccur.
10Onimaging,hemothoraxandhemomediastinumarethemostcommonndings1;CTangiographyistheprimarynoninvasivediagnosticmodalityforBAAs.
11However,conventionalangiographycanbeperformedforbothdiagnosticandtreatmentpurposes.
5Nowadays,endo-vasculartechniquessuchastranscatheterembolizationhasbeenincreasinglyapplied.
Insummary,herewereportedararecaseofrupturedBAA,whichhasnotbeendescribedbefore.
CorrectdiagnosisofthisproblemcanbeobtainedbyCTangiographyandselectivearteriography.
ThispatienthadahistoryofDVT.
Therefore,whenhesuddenlyhadabdominalpain,wespontaneouslyassumedthathehadpulmonaryembolism,whileignoringothercauses.
Ifthepatienthadcontinuallytakenanticoagu-lantantithromboticdrugs,avirulentbleedingwouldhaveFig.
1(A)TherewasnomediastinalbleedingintheCTscanofthechestwhenthepatientwasadmitted.
(B)TherewasamediastinalbleedingintheCTscanafterthepatientexperiencedsuddenupperabdomenpain.
(D–F)AorticarteryCTdemonstratedaruptureontherightbronchusartery.
(G)Aselectivearteriographyfurtherconrmedthattherewasanacutebilateralhemothoraxsecondarytoarupturedbronchialarteryaneurysmoftherightbronchialartery.
(H)Thebronchusarteryaneurysmwassuccessfullytreatedbycoilembolization.
ThoracicandCardiovascularSurgeonReportsVol.
5No.
1/2016AnticoagulantAgent–InducedRuptureofaMediastinalBAAWangetal.
19occurred.
Takentogether,commonpresentationischestpain,whichmayleadtoconfusionwithothersyndromes,suchaschestwalldiseasesanddigestivesystemdiseases,soCTangiographyisnecessaryfordifferentialdiagnosisofchestpainandabdominalpain.
ConictofInterestTheauthorsdeclarethatthereisnoconictofinterest.
FundingThisworkwasnotsupportedbyanyfunding.
Reference1SeoYH,KwakJY.
Spontaneoushemomediastinumandhemo-thoraxcausedbyarupturedbronchialarteryaneurysm.
KoreanJThoracCardiovascSurg2011;44(4):314–3172TailléC,FartoukhM,HoulR,KobeiterH,RémyP,LemaireF.
Spontaneoushemomediastinumcomplicatingsteroid-inducedmediastinallipomatosis.
Chest2001;120(1):311–3133YokoyamaH,OhmiM,SadahiroM,ShojiY,TabayashiK,MoizumiY.
Spontaneousruptureofthethoracicaorta.
AnnThoracSurg2000;70(2):683–6894MatsugeS,HosokawaY,MurakamiY,SatohK.
[Thymomawithidiopathicthrombocytopenicpurpuraprecededbymediastinalhemorrhage;reportofacase].
KyobuGeka2002;55(10):899–9025MizuguchiS,InoueK,KidaA,etal.
Rupturedbronchialarteryaneurysmassociatedwithbronchiectasis:acasereport.
AnnThoracCardiovascSurg2009;15(2):115–1186PugnaleM,PortierF,LamarreA,etal.
Hemomediastinumcausedbyruptureofabronchialarteryaneurysm:successfultreatmentbyembolizationwithN-butyl-2-cyanoacrylate.
JVascIntervRadiol2001;12(11):1351–13527VosseBA,vanBelleAF,deVriesGJ,DasM.
Hemomediastinumduetospontaneousruptureofamediastinalbronchialarteryaneu-rysm—ararecauseofthoracicpain.
RespirMedCaseRep2014;12:27–298ShihSY,HuSY,TsanYT,LinTC.
Spontaneoushemomediastinumandhemothoraxcausedbyrupturedbronchialarteryaneurysm.
EurJCardiothoracSurg2009;35(6):10769ChatterjeeA,GhoshS,SalhiyyahK,GainesP,RoccoG.
Ararepresentationofarupturedbronchialarteryaneurysm.
Thorax2004;59(10):91210WilsonSR,WingerDI,KatzDS.
CTvisualizationofmediastinalbronchialarteryaneurysm.
AJRAmJRoentgenol2006;187(5):W544-511HoffmannV,YsebaertD,DeSchepperA,ColpaertC,JorensP.
Acutesuperiorvenacavaobstructionafterruptureofabronchialarteryaneurysm.
Chest1996;110(5):1356–1358ThoracicandCardiovascularSurgeonReportsVol.
5No.
1/2016AnticoagulantAgent–InducedRuptureofaMediastinalBAAWangetal.
20
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