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Clinical Medical & Case Reports
Open Journal of
ISSN2379-1039
Volume3(2017)Issue14
YigitbasBA
OpenJClinMedCaseRep:Volume3(2017)
AnorexianervosaaccompaniedbybilateralpneumothoracesAybukeKekecoglu,MD;BurcuArpinarYigitbas,MD*;FadimeKelesoglu,MD;BarisSeker,MD;AyseF
Kosar,MD
*BurcuArpinarYigitbas,MD
I�ntensiveCareUnit,YedikuleChestDiseaseandSurgeryTrainingandResearchHospital,Istanbul,
Turkey.Phone:00904090202;Email:[email protected]
Abstract
Anorexianervosaisaneatingdisorderthatcanbeaccompaniedbyvariouscomplications.An18-year-
oldfemalepatient,whowastreatedfor2yearsduetoanorexianervosa,wasadmittedtoourhospital
because of bilateral pneumothoraces. A bilateral tube thoracostomy was performed, and she was
diagnosedwithidiopathicspontaneouspneumothoraxanddischarged.Thepatientwasadmittedtothe
hospital 9 months later due to respiratory complications. No increase was found in the patient's
pneumothorax, but hypercarbic respiratory failure was discovered. During her treartment in our
intensivecareunitnoneofhercultureswerepositive.Thepatientwasundernon-invasivemechanical
ventilationandmonitored.Shewasintubatedfollowinganincreaseinparenchymalin�iltratesandinthe
severityofherconcurrent respiratory failure.Thepatientdieddue toARDSon the17thdayofher
hospitalization.
Keywords
acuterespiratorydistresssyndrome;anorexianervosa;bilateralpneumothoraces
Introduction
Eatingdisordersarebecomingincreasinglyprevalentinoutpatientclinics.Thesedisordersmay
affecteveryorganandsysteminthebody;however,thelungsaretheless-commonlyaffectedorgans.In
thesecases,spontaneouspneumothorax,pneumomediastinumandemphysemaarereportedtodevelop
frequently[1].Inthisparticularcasestudy,thepatienttobediscussedwastreatedfor2yearsforthe
diagnosis of anorexia nervosa, a tube thoracostomy was performed following the development of
bilateral pneumothoraces, but the patient was transferred to the intensive care unit due to the
deteriorationofhergeneralmedicalcondition.
CasePresentation
The18-year-oldfemalepatientwasadmittedtoemergencyroomcomplainingofshortnessof
breath,respiratorydisorder,andcoughaccompaniedbyexpectoration.Apostero-anteriorchestX-ray
showedbilateralpneumothoracesaccompaniedbyin�iltrationsconcordanttopneumonia. Thepatient
washospitalizedinthechestdiseasesclinic,butthenreferredtotheintensivecareunitwhenhergeneral
medicalconditiondeterioratedandherhypoxemicrespiratoryfailureworsenedinarterialbloodgas.
Accordingtothepatient'shistory,shehadbeentreatedfor2years bythepsychiatrydepartment dueto
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herdiagnosisofanorexianervosa.Shewasanonsmoker.
The patient �irst admitted to emergency room approximately 9months ago, complaining of
shortnessofbreathandcough.Shewashospitalizedduetopneumothoraxobservedinbothofherlungs
during examinations. The case was discussed with the chest surgery department and a tube
thoracostomywasperformed(Fig1).Drainagewasterminatedwhennoincreasewasobservedinthe
currentpneumothorax inboth lungsof thepatient (Fig 2).Thoracic computed tomographyshowed
bilateralpneumothoraces,whichstartedattheupperlobeapicalsegmentlevelinbothhemithoraxesand
continuedtothesuperiorlingulasegmentlevelontheleft,andwhichscatteredtothediaphragmatic
region in the rightmiddle lobe. (Fig 3,4), In the transbronchial biopsy obtained through �iberoptic
bronchoscopy (FOB), minimal �ibrotic thickening, edema and bronchial mucosa fragments were
observed in the normal alveolar septa. Eating disorder-related pneumothorax was considered; the
patientwasdischargedandreceivedoutpatienttreatment.Duringfollow-uptreatments,noprogression
wasobservedinthepneumothorax,whichwasobservedintheupperzonesofbothlungs.
9monthslaterwhenshereadmittedtoouremergencyroomandacceptedtoourrespiratory
intensivecareunit,hergeneralmedicalconditionwaspoorandshewasconfused,dyspneic,tachypneic
andtachycardic.Herappearancewasquitecachecticandbodymassindexwas13kg/m2.Duringthe
physicalexamination,itwasobservedthatrespiratorysoundswerereducedintheupperareasofboth
lungs, and her expiration was longer. Bilateral and common coarse crackles were present during
inspiration. From the cardiovascular system examination S1(+), S2(+), the following results were
obtained:pulse:142/min,arterialbloodpressure:100/60mmHg,arterialoxygensaturationinroomair:
87%,arterialbloodgasvaluesinroomair:Ph:7.29,PCO2:86mmHg,PO2:60mmHg,HCO3:37mmol/L,
PaO2/FiO2:285.Thepatientwasgivennon-invasivemechanicalventilationunderBIPAPvision.IPAP26
cmH2O,EPAP6cmH2OandFiO2werestartedwith35%.Biochemistryanalyseswereasfollows:Glucose
68mg/dl;Urea22mg/dl;Creatinine0.3mg/dl;Uricacid1.6mg/dl;Totalprotein6.6g/dl,2.8g/dl;
Calcium9.2mg/dl;Sodium134mmol/L;Potassium4.5mmol/L;Chlorine98mmol/L;Totalbilirubin0.7
mg/dl;AST16U/L;ALT11U/L;GGT56U/L;LDH190U/L;ALP141U/L;totalcholesterol71mg/dl;
triglyceride78mg/dl;HDL16mg/dl;LDL39mg/dl;VLDL16mg/dl;Iron4mmg/dl;UIBC156mmg/dl;
CRP:33mg/L;WBC:14.9103/mm3;neutrophile10.5103/mm3;haemoglobin13.7g/dl;haematocrit
41.3%.Inthepostero-anteriorchestX-ray,itwasobservedthatthepneumothoraxlinewhichwaslimited
to the bilateral apical region had extended, and that common in�iltrations and consolidation had
developed in the lower right zonewhen compared to theX-ray from9months ago (Fig 5). Thorax
computedtomographyshowedpneumothoraxandcommonperibronchial in�iltrationswithbilateral
ground-glassdensitywerepresent inboth lungs,andnon-homogeneousconsolidation includingair
bronchogramswerepresentinthelaterobasalandporterobasalsegmentsoftherightlung'slowerlobe
(Fig6,7).Thepatient'sclinicalconditiondeterioratedandconfusionincreased,andthefollowingresults
wereobtainedfromarterialbloodanalysis:Ph:7.02,PCO2:129mmHg,PO2:84mmHg,HCO3:33.5
mmol/L,satO2:87.5%FiO2:100%,PaO2/FiO2:84.Shewasthenintubatedbasedontheseresults.Inthe
pressurecontrolmode,shewassupportedwithPEEP:8cmH2O,AbovePEEP:24cmH2OFiO2:40%,
respiratoryrate14.Inherechocardiography,apericardialeffusionof9.5mmwasdetectedintheleft
ventricleposterior,whichsurroundedtheheartentirely.Herejectionfractionwasmeasuredas60–62%.
Intheetiologicalanalyses,immuneglobulinlevelswerenormal,P-ANCAandC-ANCAwerenegative,and
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negative, and rheumatoid level was normal. Blood, urine or deep tracheal aspirates cultures were
negative.Sputumandlavagewerenegativeintermsofacidfastbacilli.Thepatient'sclinicalcondition
deteriorated rapidly, bilateral in�iltrations increased (I�mage 8), ARDS and shock developed and
respiratoryfailureworsened;asaresult,multi-organfailuredeveloped.Thepatientdidn'trespondto
interventionsanddiedonthe17thdayofherhospitalization.
Discussion
Anorexianervosaisaneatingdisorderthatcanbeaccompaniedbyvariouscomplications.Ithas
beenreportedtoaffectgastrointestinal,cardiovascular,hematologic,endocrine,renal,neurologicaland
dermatologicalsystems[1].Therespiratorysystemmaybeaffectedaswell,butitspathophysiologyisnot
yetde�inite.Inanimals,ithasbeendeterminedthatlong-termfastingleadstoadecreaseintotallung
protein,connectivetissue,hydroxyprolineandelastinlevels[2].Emphysemaisalsobelieveddevelopin
thismanner,butthisdamageisde�inedasemphysema-likeratherthanemphysema.
After administering tube thoracostomy to the patient who developed bilateral spontaneous
pneumothorax,clinicalrecoverywasachievedbuttotalexpansionwasn'tobtainedradiologically.The
patientwasconsultedbythechestsurgeryteam,tubethoracostomywasendedandthepatientreceived
outpatient follow-up treatment. Following discharge, the patient was admitted for two follow-ups
treatments at one-month intervals. It was observed that she had no complaints and that the
pneumothorax line hadn't extended. The patient didn't attend the subsequent follow-ups, but was
admittedtotheintensivecareunitinthe9thmonthduetodeteriorationinhergeneralmedicalcondition
anddevelopmentofrespiratoryfailureeventhoughthepneumothorax linehadn'textended.Thorax
computed tomography showed no increase in bilateral pneumothorax, whereas bronchiectasis and
�ibrosishaddevelopedaswellasconsolidation.Nopathologywasobservedinthepatientthatwouldlead
toapicalbullaorpneumothorax.Thepatient'sconditionwasconsideredassecondarytomalnutrition,
andassociatedwithherrecovery'sdelayandprevention.Eventhoughanutritionprogram(intravenous
liquid,bloodproducts,parenteralandenteralnutrition)wasinitiatedforthepatient,noimprovement
wasobservedinthegeneralconditionandoxygenationparameters.Bif�letal.statedthattreatmentwith
VATS was successful following the long-term leak in the patient with anorexia nervosa and
pneumothorax;asmentioned,structuralchangesoccurinthelungofpatientswithanorexianervosaand
thusleadtoadecreaseinsurfactantgrowthandalimitedrecoveryofthelungs[1].Corlessetal.stated
that bilateral tube thoracostomy was performed in patients with anorexia nervosa and bilateral
spontaneouspneumothorax,andthatabilateralthoracoscopicpleurectomywasperformedtoprevent
recurrence. In patients with anorexia nervosa, alveolar ruptures are thought to emerge from any
condition (such as coughing, vomiting) in which the alveolar structure weakens and intrathoracic
pressure increases [2]. In a case of anorexia nervosa reported byDanzer et al., and also Lee et al.,
pneumomediastinum, pneumothorax and subcutaneous emphysema were observed.
Pneumomediastinum, pneumothorax and subcutaneous emphysema are rarely seen together. It is
thoughtthatthepatient'svomitinganamnesiscausedtheintraabdominalandintrathoracicpressuresto
increaseandtheconditiontooccurevenifthepatientisnotvomitting[3,4].Hochlehnertetal.reporteda
patient with anorexia nervosa who developed spontaneous pneumothorax, and highlighted that
anorexia nervosa caused pneumomediastinummore commonly than pneumothorax. This is a rare
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condition that is generally self limited; its treatment lies in providing analgesia in addition to
conservative treatment [5]. Malnutrition is reported to be associated with emphysema, bulla and
bronchiectasisdevelopment,andthereisacorrelationbetweenBMIandtomographymeasurementof
emphysema[6].Inpatientswithanorexianervosa,itisbelievedthatperipheralintrapulmonaryairleaks
progressinaretrogradefashion:theairisdissectedintheinterstitialareaalongthebronchialtree,and
eventually leads to wide mediastinal, retroperitoneal and subcutaneous emphysema even without
smoking,becauseofthisdif�isioncapacityforoxygendecreases[7].Generally,supplementaltreatment
andweightgainareexpectedtoleadtospontaneousrecoveryinmostcases.
Limitedandlittlerecoveryofourpatientledtodeteriorationofhergeneralmedicalcondition,and
deteriorationofhergeneralmedicalconditionledtodecreasedrecovery.Thepatient,whosepulmonary
functions decreased over time and whose respiratory muscles appeared to have weakened, was
intubated.InthecontrolchestX-ray,consolidationfromhilustoperipherywasdetectedinbothlungs,
anditwasbelievedthatsurfactantde�iciency-relatedARDShaddevelopedinthepatient.Thepatient
didn'trespondtotreatmentanddied.
Conclusion
Anorexianervosacanaffectallsystemsinthebody,includingtherespiratorysystemandlungs.
Spontaneouspneumothorax isoneof thecomplicationsassociatedwiththisdisease.Long-termand
severemalnutritionmaylimitrecoveryandevenmakeitirreversible.Emphysema-likedestructionand
surfactantde�iciencywerereportedinthesepatients.Asinourcase,thegeneralmedicalconditionmay
deteriorate, the damagemay be irreversible, and the patientmay die. Such complicationsmust be
considered inpatientswith anorexianervosa, and earlynutrition and treatmentprogramsmust be
initiated.
Figures
Figure1:ChestX-rayofthepatienttakenduring�irst
hospitalizationshowingbilateralpneumothoracesand
p e r f o rmed b i l a t e r a l t u b e t h o r a c o s t omy.
Pneumothoracesareshownbyboldarrows.
Figure2:ChestX-rayof thepatientat the timeof
drainremoval,pneumothoracesinbothlungsdidn't
resolve completely but there was no increase in
pneumothoraces. Pneumothoraces are shown by
boldarrows.
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Figure 3,4: Chest computed tomography taken during the patient's �irst hospitalization showing bilateral
pneumothoraces.Pneumothoracesareshownbyarrows.
Figure5:ChestX-rayofthepatienttakenthird
day in the respiratory intensive care unit in
secondhospitalizationafter9monthsshowing
bilateralpneumothoracesprogressionandnew
in�iltrations.
Figure 6,7:Chest computed tomography of the patient 15th day in the
respiratory intensive care showing pneumothorax and in�iltrations with
bilateralground-glassdensitywithnon-homogeneousconsolidationinthe
rightlung'slowerlobe
Figure 8: Chest X-ray of the patient taken 15th day in the respiratory
intensivecareunitshowingbilateralin�iltrationsresultedinARDS2days
beforepatientwasdeceased.
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References
1.Bif�lWL,NarayananV,GaudianiJL,MehlerPS.Themanagementofpneumothoraxinpatientswithanorexia
nervosa:Acasereportandreviewoftheliterature.PatientSafSurg.2010;4(1):1.
2.CorlessJA,DelaneyJC,PageRD.Simultaneousbilateralspontaneouspneumothoracesinayoungwomanwith
anorexianervosa.I�ntJEatDisord.2001;30(1):110–2.
3.DanzerG,MulzerJ,WeberG,LembkeA,KocaleventR,KlappBF.Advancedanorexianervosa,associatedwith
pneumomediastinum,pneumothoraxandsofttissueemphysemawithoutesophageallesion.I�ntJEatDisord2005;
38(3):281–284.
4.LeeKJ,YumHK,ParkIN.SpontaneousPneumomediastinum:AnUnusualPulmonaryComplicationinAnorexia
Nervosa.TubercRespirDis2015;78(4):360–362.
5.HochlehnertA,LoweB,BludauHB,BorstM,ZipfelS,HerzogW.Spontaneouspneumomediastinuminanorexia
nervosa:Acasereportandreviewoftheliteratureonpneumomediastinumandpneumothorax.EurEatDisorders
Rev2010;18:107–115.
6.CoxsonHO,ChanIHT,MayoJR,HlynskyJ,NakanoY,BirminghamCL.Earlyemphysemainpatientswithanorexia
nervosa.AmJRespCritCareMed2004;170:748–752.
7.MehlerPS:BrownC.AnorexiaNervosa–medicalcomplications.JEatDisord2015;3:11.
ManuscriptInformation:Received:March02,2017;Accepted:July04,2017;Published:July10,2017
1 2 2 2AuthorsInformation:AybukeKekecoglu,MD ;BurcuArpinarYigitbas,Md *;FadimeKelesoglu,MD ;BarisSeker,MD ;AyseF2Kosar,MD
1I�ntensiveCareUnit,YedikuleChestDiseaseandSurgeryTrainingandResearchHospital,Istanbul,Turkey2DepartmentofChestDiseases,YedikuleChestDiseaseandSurgeryTrainingandResearchHospital,Istanbul,Turkey
Citation: Kekecoglu A, Yigitbas BA, Kelesoglu F, Seker B, Koasar AF. Anorexia nervosa accompanied by bilateral
pneumothoraces.OpenJClinMedCaseRep.2017;1285
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