Necrotizing pneumonia with bronchopleural fistula as ... - iKnow

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Case Report

Necrotizing pneumonia with bronchopleural fistula as an uncommon complication of pneumonia in children: a case report ^✩

Damayanti Sekarsari, MD

^a^,^∗

, Syeida Handoyo, MD

^a

, Mohamad Yanuar Amal, MD, MBA

^a

, Primadea Kharismarini, MD

^b

aDepartmentofRadiology,Dr.CiptoMangunkusumoNationalGeneralHospital,FacultyofMedicine,Universitas Indonesia,Jakarta,Indonesia

bFacultyofMedicine,UniversitasIndonesia,Jakarta,Indonesia

a r t i c l e i n f o

Articlehistory:

Received17March2021 Revised4May2021 Accepted4May2021

Keywords:

Necrotizing Pneumonia

Bronchopleuralfistula Children

Imaging

a b s t r a c t

Necrotizingpneumoniaisanuncommonbutseverecomplicationofcommunityacquired pneumoniacharacterizedbythedevelopmentofnecrosis,liquefaction,andcavitationofthe lungparenchyma.Itoccursinfrequentlyinchildren,rangingfrom0.8%to7%ofcommunity acquiredpneumoniacases.Wereportedacaseof28-month-oldfemaleinfantwithahistory ofseveredyspneaandfever5daysbeforeadmission.Afteradministrationofappropriate antibioticsforpneumonia,thepatient’sconditionwasstillunresolved.Then,contrastCT scanshowedcavitarylesionswithinconsolidatedlungswithlossofvolumeandlackofcon- trastenhancementthatconfirmedthediagnosisasnecrotizingpneumonia.Thepresence ofpneumothoraxinthepatientdepictsapossiblebronchopleuralfistulawhichsignificantly increasemorbidityandmortalityrisk.Surgicalmanagementcouldnotbeimplementeddue toworseningconditionofthepatient.Itissuggestedthatpatientswithsuspicionofnecro- tizingpneumoniaaresubjectedtochestCTscantoavoiddelayindiagnosisandappropriate management.

ThisisanopenaccessarticleundertheCCBY-NC-NDlicense (http://creativecommons.org/licenses/by-nc-nd/4.0/)

Introduction

Necrotizingpneumonia(NP)isaprocess ofnecrosis,liquefaction and cavitation of the lung parenchyma caused by infectious pathogen [1]. It is characterized by a condition of progressive pneumonia in previously healthy children

✩ CompetingInterests:None

∗ Correspondingauthor.

E-mailaddress:[email protected](D.Sekarsari).

despite administration of adequate antibiotic treatment [2,3].Occurrenceofbronchopleuralfistulacanfurthercause deterioration ofthe patient. NP occurs in 3.7%ofcommunity pneumonia. However, retrospective studies show an increasingincidenceinthelast20years[1].

ThediagnosisofNPisdeterminedbyusingimagingmodal- itiesthatshowmultiplethin-walledcavitieswithinthecon-

https://doi.org/10.1016/j.radcr.2021.05.008

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Fig.1– Chestradiographyperformedonthe1stdayof hospitalizationshowshomogeneousopacityintheright hemithoraxwithinfiltratesintheupper-middlefieldofthe leftlung.

solidationareaofthelung.Pathologicalexaminationofau- topsy or resection of lung specimens reveals lung inflam- mation,alveolarconsolidationandintrapulmonaryvascular thrombosiswithnecrosisandmultiplesmallcavities[3].

CTscanwithcontrastremainsasthegoldstandardforthe diagnosis ofNP.Chestradiography havealower sensitivity comparedtoCTscanbecauseconsolidationandeffusioncan concealsmallradiolucentlesions.EarlydiagnosisofNPwill affecttheintensityofpatientsurveillance,choiceoftherapy anddurationofhospitalization.

Case report

A28-month-oldgirlcametotheEmergencyRoominDr.Cipto MangunkusumoNationalHospital(RSCM)withcomplaintsof severeshortnessofbreathandfever5daysbeforeadmission.

ThispatientwasadmittedtothehospitalbeforetheCOVID- 19pandemic.Tendaysbeforebeingadmittedtothehospital, thepatienthadcomplaintsofcough,runnynose,andconsti- pation.Oninitialphysicalexamination,hervitalsignswere blood pressure 107/60mmHg, pulse170 bpm, temperature 37.2°C,andrespiratoryrate40breathsperminute.Thereare ronchiandwheezinginbothlungs.Laboratoryexamination showedanemiawithHb5.4g/dL.Thenumberofleukocytes increased(32,380cells/mm)withaProcalcitoninvalueof0.77 ng/mLwhichindicatesepsis.

The chest radiography examination revealed a massive rightpleuraleffusion,possiblyaccompaniedbyrightlungat- electasis,andleftlunginfiltrates(Fig.1).Furthermore,thepa- tientwassubjectedtoathoracicultrasound whichshowed acomplexrightpleuraleffusionwithpleuralthickeningand suspicionoflocalizedpleuraleffusionwithrightlungconsol- idationinthemedialside(Fig.2).

Pleuralfluidtappingandwatersealeddrainage(WSD)in the patient revealed a cloudy yellow, seropurulent pleural fluid.Cytology analysis showed that the pleural fluidcon- sistedofmesothelial cells,macrophages,andlymphocytes.

Therewerenomalignanttumorcellsfoundintheanalysis.

ContrastchestCTscanwasperformedandshowedheteroge- neousconsolidationwithmultiplecavitiesin1to5segments ofthe rightlung,narrowing ofthe rightsuperiorbronchial branchwhichsuggestsNP,paraaorticwindowandsub-carina lymphadenopathy,righthydropneumothorax,andleftpleural effusion(Fig.3).

Thepatient receivedablood transfusion duetoanemia andwasadministeredampicillinsulbactam250mg/6hours, andmeropenem 400mg/8hours.Follow-up chestradiogra- phyexaminationwasperformedaweeklaterwhichshowed rightlung consolidation with multiplebullae,right middle andlowerlungatelectasis,righthydropneumothorax,andleft pleural effusion (Fig. 4). WSD production had increased to 30to 50ml.Thepatient was initiallyplanned forthoraco- tomyresectionofthebullaeandlobectomy.However,since thegeneralconditionofthepatientcontinuedtodeteriorate, thesurgerywasdelayed.

Duringhospitalization,thepatientstillhadcomplaintsof persistentcough,shortnessofbreath,andfever.Onphysical examination,thebreathsoundintherightlungisweakened andwheezingisheard.Thepatientwasgivenadditionaltreat- mentofVentolinandCombiventnebulization.Theresultsof bloodculture,IgMandIgGofrubella,NS1,sputumAFBand GeneXpertexaminationswerenegative.

Onthe16thdayoftreatment,thepatientclinicalcondition hadworsenedwithcomplaintsofsevere dyspneaandhigh feverupto40°C.Previousantibiotictreatmentswerereplaced withCefotaxime-Sulbactam250g/6hoursandAmikacinwith loadingdoseof250mg/12hours,followedby180mg/24hours.

Atthattime,thepatientwasadvisedtobetreatedatthePICU.

Thefamilydecidedtowithdrawcardiopulmonaryresuscita- tiononthepatient.Onthe20thdayoftreatment,thepatient begantoappearsomnolentwithgaspingbreath,acralcold- ness,andweakpulse.Then,thepatientexperiencedcardiac arrestandwaspronounceddead.

Discussion

Necrotizingpneumoniaisdefinedasmultiplecavitieswith- out marginal enhancement in the necrotic areas of lung parenchyma[4].Necrosisofthelungparenchymaoccursdue tothrombotic occlusion ofthe alveolarcapillaries because ofinflammation that leads to ischemia [5]. This condition israrelyseeninchildren[6,7].Pediatricpatientswithpneu- moniasymptoms,suchaspersistentfeveranddyspnea,that donotimprovedespiteadministrationofadequateantibiotic treatmentneedtobeevaluatedforthediagnosisofNP[8].

CTscanremainsasthegoldstandardforthediagnosisof NP.ChestradiographyislesssensitivethanCTscansbecause consolidationandeffusioncanconcealsmallradiolucentle- sions[4].Furthermore,CTscanissuperiorinassessingfurther complicationsofthelungparenchymaandpleura [9].Diag- nosticfindingsonCTscanincludelossofnormalparenchy-

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Fig.2– Ultrasonographyperformedonthe1stdayofhospitalizationrevealpleuraleffusionwithinternalecho,pleural thickeningacrosstherighthemithoraxwithcollapsedlung,andconsolidationintheupperlobeoftherightlung.

Fig.3– CTscanperformedonthe1stdayofhospitalizationshowsheterogeneousconsolidationintherightlung(A), bilateralpleuraleffusionwithairinthepleuralspaceoftherighthemithoraxwithpartialcollapseoftheinferiorlobeofthe rightlung(BandC).Narrowingoftherightsuperiorbronchialbranchisnoted(B).Thereisalsofibrosisandthickeningof themultipleinterceptionsinthemedialandinferiorlobesoftherightlung(D).

malpattern,decreasedparenchymalenhancement,andmul- tiplethin-walledcavities[4,10].

Inthispatient,consolidationandcavitieswithoutcontrast enhancementonchestCTscanwereconsistentwithNP.Inad- dition,multiplebullae,pleuraleffusions,andlocalizedpneu- mothoraxwereshowninCTscan,indicatingcomplicationsof

NP.Thepresenceofapneumothoraxdepictsapossiblebron- chopleuralfistula.Inanotherstudy,bronchopleuralfistulaoc- curredin63%ofpatientsand80%ofpatientsrequiredsurgical therapy[10].

Themost common causative microorganismsin NP are StaphylococcusaureusandStreptococcuspneumonia.How-

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Fig.4– FollowupexaminationofchestradiographywithAPandlateralprojectionswereperformedonthe8thdayof hospitalization,showingmultiplebullaeandhydropneumothoraxwithcollapsedlungintherighthemithorax,and consolidationwithmultiplecavitiesintheupperlobeoftherightlung.

ever,causativemicroorganismsare rarelydetectedand can onlybefoundinhalfofthecases[11].Inthispatient,there were nomicroorganisms foundonblood cultureor pleural fluidanalysis.Thisresultcanbeinfluencedbytheadminis- trationofempiricantibioticstothepatient.

The patient’s worsening condition of fever, prolonged shortnessofbreath,andsuspectedbronchopleuralfistulacan beanindicationforsurgicalmanagement.However,thereis noclearconsensusregardingthesurgicalmanagementofNP.

Inthispatient,surgicalmanagementwasplannedforbullae resectionandpossiblypneumonectomy,butthepatient’scon- ditioncontinuedtodeteriorateandeventuallydiedbeforesur- gicalmanagementwasimplemented.Althoughtheprognosis ofNPisgenerallygood,complicationssuchasbronchopleural fistulainthepatientcanheavilyimpacttheoutcomeofthe patient[7].

Conclusion

Necrotizingpneumoniaisone oftherare complicationsof pneumoniainchildrenthatpresentsseveremorbidity.NPis characterizedbyfeverandprolongedshortnessofbreaththat doesnotrespondtoadequate antibiotictreatment.Thedi- agnosisisconfirmedbyfindingaconsolidationwithmulti- plecavitiesonchestCTscanexamination.Whenpatientsare suspectedwithNP,chestCTscanexaminationisnecessaryto avoiddelayindiagnosisandappropriatemanagement.

Patient consent

Alongwiththisletter,wewouldliketoconfirmthatourpa- tienthasagreedthatherdaughter’smedicalhistorycanbe publishedasacasereportpaper.Inordertoprotectthepa- tient’sprivacy,wedidnotincludethephysicalappearanceof

thepatient.Furthermore,wefocusedonimagingandproof ofsurgicaloutcomesofthepatient,sowedidnotviolateany patient’sprivacy.

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