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ส§¢ÅÒ¹¤ÃÔ¹·Ã์àǪสÒà »‚·Õè 33 ©ºÑº·Õè 2 มÕ.¤.-àม.ย. 2558 Songkla Med J Vol. 33 No. 2 Mar-Apr 2015
Case Rep
ort
103
64 MDCT 3D Assessment of Complex Congenital Heart and Bilateral Tracheal Bronchi: A Case Report
ศุภÔกÒ กฤษณÕไพºูÅย์1*สุภÒพÃ โÃยมณÕ2
64 MDCT 3D Assessment of Complex Congenital Heart and Bilateral Tracheal Bronchi: A Case Report.Supika Kritsaneepaiboon1, Supaporn Roymanee2
1Department of Radiology, 2Department of Pediatrics, Faculty of Medicine, Prince of Songkla University, Hat Yai, 90110, Thailand.*E-mail: supikak@yahoo.comSongkla Med J 2015;32(2):103-107
º·¤Ñดย่อ: ภาวะแขนงหลอดลมงอกจากท่อลมใหญ่เป็นส่วนหนึ่งของความผิดปกติแต่กำาเนิดของหลอดลมและท่อลมใหญ่ นอกจากนั้นภาวะแขนงหลอดลมงอกจากท่อลมใหญ่ทั้งสองข้างพบได้น้อยมากและมักไม่มีอาการ รายงานผู้ป่วยเด็กหญิงอายุ8ปีมีโรคหัวใจพิการแต่กำาเนิดชนิดซับซ้อนซึ่งประกอบด้วยหัวใจห้องล่างหนึ่งห้องลิ้นหัวใจไทรคัสพิดไม่เจริญและหลอดเลือดแดงใหญ่ของปอดตีบอย่างมากได้พบภาวะแขนงหลอดลมงอกจากท่อลมใหญ่ทั้งสองข้างและภาวะกลุ่มอาการไม่มีม้ามอย่างบังเอิญจากการตรวจโดยเครื่องเอกซเรย์คอมพิวเตอร์และฉีดสารทึบรังสี สûุ:ภาวะแขนงหลอดลมงอกจากทอ่ลมใหญท่ัง้สองขา้งสามารถพบไดใ้นผูป้ว่ยภาวะกลุม่อาการไมม่มีา้มและเชื่อว่าเป็นแขนงหลอดลมข้างขวาสองข้าง
¤ำÒสำÒ¤Ñญ:กลุม่อาการทีไ่มส่ามารถแยกซา้ย-ขวาได,้กลุม่อาการไมม่มีา้ม,ภาวะแขนงหลอดลมงอกจากทอ่ลมใหญ่
1ภÒ¤ÇÔªÒÃѧสÕÇÔ·ยÒ 2ภÒ¤ÇÔªÒกุมÒÃàǪศÒสตÃ์ ¤ณะแพ·ยศÒสตÃ์ มหÒÇÔ·ยÒÅÑยส§¢ÅÒ¹¤ÃÔ¹·Ã์ อ.หÒดใหญ่ จ.ส§¢ÅÒ 90110ÃѺต้¹©ºÑºÇѹ·Õè 16 มกÃÒ¤ม 2558 ÃѺŧตÕพÔมพ์Çѹ·Õè 2 àมษÒย¹ 2558
ส§¢ÅÒ¹¤ÃÔ¹·Ã์àǪสÒà »‚·Õè 33 ©ºÑº·Õè 2 มÕ.¤.-àม.ย. 2558 104
โäหÑÇใจพÔกÒÃแต่กำÒà¹ÔดซѺซ้อ¹Ã่ÇมกѺภÒÇะแ¢¹§หÅอดÅม§อกจÒก·่อÅมใหญ่
ศุภÔกÒ กฤษณÕไพºูÅย์ แÅะ สุภÒพÃ โÃยมณÕ
Abstract: Thetrachealbronchusmanifestsavarietyoftracheobronchialanomalies.Bilateraltrachealbronchiarerare.Anomaloustrachealbronchusisusuallyasymptomatic. Case report: Wereportan8-yearoldgirlwhohadunderlyingcomplexcongenitalheartdiseases:uni-ventricularheart,tricuspidatresia,andseverepulmonarystenosisandwhowasincidentallyfoundtohaveaspleniasyndromeandbilateraltrachealbronchifromthecontrastenhancedmultidetectorcomputedtomography(MDCT). Conclusion:Thebilateraltrachealbronchiaremoreoftenfoundinpatientswithaspleniasyndromeandconsideredbilateralrightbronchi.
Keywords:asplenia,heterotaxysyndrome,trachealbronchus
Introduction Trachealbronchusisrelativelyrareandoftenincidentallydiscoveredduringbronchoscopeoratchestcomputedtomography(CT).1-3Thepresen-tationcanvaryfromasymptomaticclinicalwithconservativetreatmentorrespiratoryproblemsinintubatedpatients.2-4Trachealbronchusismorecommoninpatientwithassociatedconditionsuchascongenitalheartdiseasethaninpatientswithoutcongenitalheartdisease.2Thiscasepresentedbilateraltrachealbronchiwithheterotaxysyndrome.
Case report An8-yearoldgirlpresentedwithunderlyingcomplexcongenitalheartdiseases:uni-ventricularheart,tricuspidatresia,andseverepulmonarystenosis.ThepatientunderwentbilateralBlalockTaussigshuntandthenbilateralGlennoperationat1yearofageandlateralFontanoperationat7yearsofage.Shehadnohistoryofrecurrentpneumonia.
ThechestCTwasperformedtoevaluatecausesofdesaturation.ArterialandvenousenhancedCTscanofthechest(PhillipsBrilliance64slice,protocol:110mAs,120kVp,2mm,40mLofUltravist370)showedtricuspidatresia,rightsidedaorticarch,severesupra-valvularpulmonarystenosis,multiplesmallaorto-pulmonarycollateralsandhepaticcongestion(Figures1Aand1B).ThelateralFontanSVCandIVCpathwaysshowednothrombusorstenosis(Figures2Aand2B)indelayedscanimages.Aspleniasyndromeandbilateraltrachealbronchi(displacedapicalsubtype)wereincidentallyfoundwithnoassociatedatelectasis(Figures3A,3Band3C).
Discussion Atruetrachealbronchusistheupperlobebronchusarisingfromthelateralwallofthetracheausuallywithin2cmandupto6cmfromthecarinaanditcouldbetiny,stenoticorblind-ended.The
Songkla Med J Vol. 33 No. 2 Mar-Apr 2015 105
Complex Congenital Heart Disease and Bilateral Tracheal Bronchi
Kritsaneepaiboon S and Roymanee S.
incidenceofrightandlefttrachealbronchusis0.1%-2%and0.3%-1%respectively.1,2Thetracheal
bronchusisclassifiedintodisplacedorsuper-
numerarysubtype.Itisdefinedasdisplacedtype
ifonebranchoftheupperlobebronchusismissing
anddescribedassupernumerarysubtypewhenthe
upperlobebronchushasanormalandcomplete
trifurcation.Thedisplacedsubtypeismorecommon
thansupernumerarysubtype.1,5Anomaloustracheal
bronchusisusuallyasymptomatic;however,recurrent
pneumonia,bronchiectasis,focalemphysema,
persistentrightupperlobeatelectasisinintubated
patientsorrespiratorydistresscanoccur,which
resultingfromimpaireddrainageattheoriginof
trachealbronchus.1,2,5Theanglebetweenthetracheal
bronchusandtracheacanvaryfrom22°to108°.
Childrenwithgreater-angletrachealbronchusor
small-sizedtrachealbronchushavelesschanceof
accidentalendotrachealintubutionthanthesmall-
angletrachealbronchustooccludethelumenof
trachealbronchusleadingtoobstructedsegmental
airwayanddeficientventilationoftheremaining
lung.2AssociatedanomaliesincludeDown’ssyndrome,
ribabnormalities,VATERsyndrome,congenital
heartdiseases,abnormalpulmonarylobulation,and
anomalouspulmonaryarterialsupplyandvenous
drainage.4-6Althoughtruebilateraltrachealbronchi
areextremelyrare,ithasbeenreportedupto24%
ofpatientswithaspleniasyndrome.7Patientswith
heterotaxysyndrome,asplenicsubtype,havetwo
“rightsides”andabsentspleen.Thebronchialpattern
inaspleniasyndromeisbilateralrightsidedbronchi
orbilateraleparterialbronchi,thusthebilateral
trachealbronchiarebelievedtobebilateralright
bronchi.2,7MDCTisthenon-invasiveandreliable
imagingtechniquetodiagnosetrachealbronchus
andalsoassociatedotheranomalies,particularly
forthreedimensional(3D)assessment.7,8The
sensitivityofMDCTtodiagnosetrachealbronchus
is100%andcanbeassistivetodetectorexclude
thisairwayabnormality.7Furthermore,theMDCT
can provide the distance between the tracheal
bronchusandcarina,thesizeoftrachealbronchus,
theanglebetweenthetrachealbronchusandthe
trachea,andparticularity3D-MDCTinparticularly
candynamicallydemonstratethespecialrelation-
shipbetweenarterialandtrachealanomalies.7,8
Figure 1A AxialenhancedMDCTshowstricuspid atresia(whitearrow)seenashypodense fattytissuepreventingconnection betweenrightatriumandhypoplastic rightventricle(*).Rightsidedaortic arch(grayarrow)isalsodemonstrated.
ส§¢ÅÒ¹¤ÃÔ¹·Ã์àǪสÒà »‚·Õè 33 ©ºÑº·Õè 2 มÕ.¤.-àม.ย. 2558 106
โäหÑÇใจพÔกÒÃแต่กำÒà¹ÔดซѺซ้อ¹Ã่ÇมกѺภÒÇะแ¢¹§หÅอดÅม§อกจÒก·่อÅมใหญ่
ศุภÔกÒ กฤษณÕไพºูÅย์ แÅะ สุภÒพÃ โÃยมณÕ
Figure 2A Coronalmulti-planarreformation (MPR)enhancedMDCTshowinglateral Fontantunnel(*)withpatentsuperior venacava(SVC)(arrow)pathway.
Figure 1B MDCT-3Dvolumerenderingview ofcardiacandgreatvesselsshowing multiplesmallmajoraortopulmonary collateralarteries(MAPCAs)(arrows) arisingfromtheproximaldescending thoracicaorta.
Figure 3A Coronalminimal-intensityprojection (minIP)imagerevealsbilateraltracheal bronchi(displacedapicalsubtype) (arrows).
Figure 2B CoronalMPRenhancedMDCTshowing lateralFontantunnel(*)withpatent inferiorvenacava(IVC)(arrow) pathway.Thespleenisabsentatleft upperabdomen(openarrow)indicating aspleniaorrightisomerism.
Songkla Med J Vol. 33 No. 2 Mar-Apr 2015 107
Complex Congenital Heart Disease and Bilateral Tracheal Bronchi
Kritsaneepaiboon S and Roymanee S.
Figure 3CCoronalMPRMDCT(lungwindow) displayingbilateralminorfissures indicatingbilateraltrilobedlungs(arrows) inrightisomerism.
Figure 3B 3D-shadedsurfacedisplay(SSD)opaque lungimagedisplayingbilateraltracheal bronchi(arrows).
Conclusion The bilateral tracheal bronchi are more
often found in patients with congenital heart
disease and asplenia syndrome considered to be
bilateral right bronchi. 3D-MDCT is the reliable,
noninvasive and powerful technique to demon-
strate associated arterial and tracheal anomalies.
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