Original Article Thirteen Years’ Experience of ...
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www.mjms.usm.my © Penerbit Universiti Sains Malaysia, 2011 For permission, please email:[email protected] Abstract Background: Diaphragmatic hernia is migration of abdominal viscera into the thoracic cavity through a defect in the diaphragm. In children, it is mostly congenital; traumatic diaphragmatic hernia being less common. This study aimed to review our experience with traumatic diaphragmatic rupture (TDR) and to identify the clinical findings and diagnostic modality that may help in early diagnosis and prompt therapy. Methods: The study involved 11 children (1–18 years old) with TDR who were hospitalised between 1993 and 2005. In addition to clinical examination, a plain X-ray of the chest and abdomen, an ultrasound, barium studies, and a computerised tomography (CT) scan were used to evaluate the patients. Results: All of the diaphragmatic ruptures occurred on the left side, with 10 occurring in the posterolateral part and 1 near the oesophageal hiatus. Two of our patients presented 7 and 10 days after the injury, and 1 patient presented 1 year after the trauma. Conclusion: TDR should remain a diagnostic possibility in children. These patients are best assessed using a CT scan. New research on stem cells and tissue-engineered bioprosthetics may pave the path for better future therapies in these cases. Keywords: acute respiratory distress syndrome, child, diagnosis, diaphragmatic hernia, medical imaging, rupture, trauma Introduction Diaphragmatic rupture is an uncommon but well-recognised complication of trauma that consists of 1.0%–5.8% of admissions into a trauma unit (1). It occurs in 0.5%–8.0% of adult trauma patients (2). In paediatric patients, it is estimated to occur at a rate of 4%–6%, and its presence has been reported to indicate high impact. It is associated with other severe injuries in 44%–94% of cases (3). Traumatic diaphragmatic injuries are usually caused by blunt abdominal trauma or penetrating injuries (2); they were first described by Ambriose Paire in 1579 (4). Due to their rarity in infants and children, such injuries can be overlooked if Original Article Thirteen Years’ Experience of Diaphragmatic Injury in Children from the Post Graduate Institute of Medical Sciences (PGIMS), Rohtak, India Kamal Nain Rattan 1 , Rajat naRang 1 , Seema Rohilla 2 , Sarita Maggu 2 , Dhara B DhaulakhanDi 3 1 Department of Pediatric Surgery, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak 124 001, Haryana, India 2 Department of Radiodiagnosis and Imaging, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak 124001, Haryana, India 3 Department of Biotechnology and Molecular Medicine, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak 124 001, Haryana, India Submitted: 6 Apr 2008 Accepted: 30 Aug 2010 unsuspected. Delayed presentation can lead to life-threatening complications as a result of organ herniation and strangulation (3). Subjects and Methods The study included children (1–18 years old) with traumatic diaphragmatic rupture (TDR) who were admitted to the Department of Pediatric Surgery of Pt BD Sharma Post Graduate Institute of Medical Sciences, Rohtak, Haryana, India, between 1993 and 2005. The following information was recorded for each patient: age, gender, duration between the trauma and hospital admission, type of injury, clinical/radiological 45 Malaysian J Med Sci. Jan-Mac 2011; 18(1): 45-51
Transcript of Original Article Thirteen Years’ Experience of ...
www.mjms.usm.my © Penerbit Universiti Sains Malaysia, 2011For permission, please email:[email protected]
Abstract
Background: Diaphragmatic hernia is migration of abdominal viscera into the thoracic cavity through a defect in the diaphragm. In children, it is mostly congenital; traumatic diaphragmatic hernia being less common. This study aimed to review our experience with traumatic diaphragmatic rupture (TDR) and to identify the clinical findings and diagnostic modality that may help in early diagnosis and prompt therapy. Methods: The study involved 11 children (1–18 years old) with TDR who were hospitalised between 1993 and 2005. In addition to clinical examination, a plain X-ray of the chest and abdomen, an ultrasound, barium studies, and a computerised tomography (CT) scan were used to evaluate the patients. Results: All of the diaphragmatic ruptures occurred on the left side, with 10 occurring in the posterolateral part and 1 near the oesophageal hiatus. Two of our patients presented 7 and 10 days after the injury, and 1 patient presented 1 year after the trauma. Conclusion: TDR should remain a diagnostic possibility in children. These patients are best assessed using a CT scan. New research on stem cells and tissue-engineered bioprosthetics may pave the path for better future therapies in these cases.
Keywords: acute respiratory distress syndrome, child, diagnosis, diaphragmatic hernia, medical imaging, rupture, trauma
Introduction Diaphragmatic rupture is an uncommonbutwell-recognisedcomplicationof trauma thatconsistsof1.0%–5.8%ofadmissionsintoatraumaunit(1).Itoccursin0.5%–8.0%ofadulttraumapatients(2).Inpaediatricpatients,itisestimatedto occur at a rate of 4%–6%, and its presencehas been reported to indicate high impact. It isassociatedwithothersevereinjuriesin44%–94%ofcases(3). Traumaticdiaphragmaticinjuriesareusuallycausedbybluntabdominaltraumaorpenetratinginjuries(2);theywerefirstdescribedbyAmbriosePaire in 1579 (4). Due to their rarity in infantsand children, such injuries can be overlooked if
Original Article Thirteen Years’ Experience of Diaphragmatic Injury in Children from the Post Graduate Institute of Medical Sciences (PGIMS), Rohtak, India
Kamal Nain Rattan1, Rajat naRang1, Seema Rohilla2, Sarita Maggu2, Dhara B DhaulakhanDi3 1 Department of Pediatric Surgery, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak 124 001, Haryana, India
2Department of Radiodiagnosis and Imaging, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak
124001, Haryana, India
3 Department of Biotechnology and Molecular Medicine, Post Graduate Institute of Medical Sciences, Pt BD Sharma University of Health Sciences, Rohtak 124 001, Haryana, India
Submitted: 6Apr2008Accepted: 30Aug2010
unsuspected. Delayed presentation can lead tolife-threateningcomplicationsasaresultoforganherniationandstrangulation(3).
Subjects and Methods
The study included children (1–18 yearsold) with traumatic diaphragmatic rupture(TDR)whowereadmitted to theDepartmentofPediatricSurgeryofPtBDSharmaPostGraduateInstitute ofMedical Sciences, Rohtak,Haryana,India, between 1993 and 2005. The followinginformationwas recorded for each patient: age,gender,durationbetweenthetraumaandhospitaladmission, type of injury, clinical/radiological
45Malaysian J Med Sci. Jan-Mac 2011; 18(1): 45-51
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Malaysian J Med Sci. Jan-Mar 2011; 18(1): 45-51
findings, relevant information regardingtraumaticsite,herniatedorgans into the thorax,associatedinjuries,andpatient’soutcome.
Result
In the study duration, 11 patients weretreatedforTDRinourhospital;8boysand3girls.Their ages ranged from 1–18 years (mean 9.5years).TheTDRoccurredfollowingblunttraumain 9 cases and a penetrating injury in 2 cases(elaborated in Table 1). Road traffic accidentswerethemostcommoncause.Themostcommonclinical findings were respiratory distress andabdominalpain.Alloftherupturesoccurredontheleftside.Theruptureswerelocalisedintheposterolateralpartsin10casesandneartheoesophagealhiatusin1case.Eightofthepatientswereadmittedsoonafter the trauma, while 1 patient was admittedafter 7 days of injury. One patient was referredtoourhospitalafter10daysofinjurywithbiliouscontentsfromthethoracicdrainagesite(admittedtoaperipheralhospitalintheinitialdaysfollowingtheinjury).Thechesttubehadbeeninsertedintothestomachbecauseitwasthoughttobeacaseofhydropneumothorax. Another patient presentedwithabdominalpainafter1yearof traumawiththe small and large bowels herniated into thethoraciccavity.Thelargebowelhadstrangulatedandrequiredresectionandanastomosis.AbnormalitieswerefoundinallofthechestX-rays (Figure 1), and all included an elevatedor indistinct diaphragm or pleural effusion.Computerised tomography (CT) scan confirmeda diaphragmatic injury with herniation of theabdominalcontentsinallcases(Figure2). After adequate investigations andresuscitation, all of these cases were treatedsurgically through an abdominal approach. Thedefectwaslocalised,theabdominalcontentswerereduced, and the defect in the diaphragm wasrepaired. Post-operatively, all patients had anuneventfulrecoverywiththeexceptionof2casesofpost-operativebronchopneumonia,whichwasmanagedmedically.
Discussion
The diaphragm is a complex,musculotendinous, dome-shaped structuredividing the thoracic and abdominal cavities. Inblunt trauma, a diaphragmatic rupture occurswhen intra-abdominal forces overcome the
Figure 2: Axialnoncontrastcomputerised tomography scan of the lower chest
showingaherniated stomach in theleft thoracic cavity in an 8-year-oldboy(arrow).
Figure 1: Chest X-ray (posteroanterior view)showing a cystic structure with anair-fluid level occupying the lefthaemithorax, causing collapse oftheleftlungandaslightmediastinalshift towards the right. The lefthaemidiaphragmisnotwelloutlined(herniating stomach mimickinghydropneumothorax).
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Table 1: Age,gender,durationbetweenthetraumaandhospitaladmission,typeof injury,clinical/radiologicalfindings,siteofthetrauma,herniatedorgansintothethorax,associatedinjuriesandoutcomeofpatients.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case1:FemaleAge5
Roadsideaccident(blunt).Bruisesoverlefthypochondrium.
Hours. Respiratorydistresswith
vomiting.Bowelsoundauscultated
inchest.
Herniationofgutintolefthaemithorax.
USG Ba studies CT scan Operative FindingsMinimalfluidintheabdominalcavity.
- - Smallgutandstomachherniatedthroughdiaphragmaticrentnearoesophageal
hiatus.Mode of Injury Duration* Clinical
FindingsPlain X-rays
Case2:MaleAge12
Roadsideaccident(blunt).Bruisesoverrightiliacfossa.
Hours. Respiratorydistresswith
vomiting.Airentrywasgoodonboth
sides.
Herniationofgutintolefthaemithorax.
USG Ba studies CT scan Operative Findings- Herniated
smallgut.- Smallgutherniated
throughposterolateralrentindiaphragm.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case3:MaleAge6
Roadsideaccident(blunt).Contusionoverhypogastriumandbruisesoverleft
iliacfossa.
1year. Respiratorydistress.
Leftpyopneumothorax.
USG Ba studies CT scan Operative FindingsTubewasinthestomach.Minimal
fluidintheabdominalcavity.
- - Spleen,smallbowel,andcolonherniatedthroughposterolateralrentindiaphragm.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case4:MaleAge8
Divinginpondfromabout3mheight.
7days. Respiratorydistresswithhydropneumo-
thorax.
Biliousfluidfromleft-sideddrain.
USG Ba studies CT scan Operative Findings- - Herniated
stomachandsmallintestineinlefthaemithorax.
Stomach,largegut,andspleenherniatedthroughpostero-lateralrentindiaphragm.
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Mode of Injury Duration* Clinical Findings
Plain X-rays
Case5:MaleAge16
Stabinjuryspanninglowerchestand
upperabdomenonleftside.
1year. Severeabdominalpainandvomiting.
Opacityinleftlungbase.
USG Ba studies CT scan Operative Findings- - - Smallgutandlarge
bowelherniaswithgangreneoflargebowel;resectionanastomosisperformed.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case6:MaleAge6
Roadsideaccident(rolledover)withbruisesoverwholeofabdomenand
back.USG Ba studies CT scan Operative Findings- - - Smallbowel,large
bowel,andspleenherniasinlefthaemithorax.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case7:FemaleAge11
Fallfromabout2.5m;had
pneumothorax.Intercostaltubewasinsertedinitially;increasedairandbiliousdrainage.Patientwasthenreferredtoourinstitute.
10days. Respiratorydistress,biliousdrainagethroughintercostaltube.
Pneumothorax;leftside.
USG Ba studies CT scan Operative Findings- - - Tubewasinthe
stomach,whichwasherniatingthroughposterolateralrent.
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Mode of Injury Duration* Clinical Findings
Plain X-rays
Case8:FemaleAge15
BulletInjurywithentrywoundinlefthypochondriumandexitwoundontheloweraspectofchest
onbackside.
Soonafterinjury.
Decreasedbreathsoundsonleftside,vomitingandrespiratory
distresson3rddayofinjury.
Haemopneumothoraxonleftside.
USG Ba studies CT scan Operative Findings- - - Stomachherniated
throughposterolateralrentinthediaphragm.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case9:MaleAge6
Roadsideaccident(rolledover)withbruisesoverwholeofabdomenand
back.
Hours. Chestpain,abdominalpain.
Herniationofgutonleftside.
USG Ba studies CT scan Operative FindingsHaemoperitoneum. - - Stomachandsmall
intestineherniatedthroughposterolateralrentindiaphragm.
Mode of Injury Duration* Clinical Findings
Plain X-rays
Case10:MaleAge18
Roadsideaccident(blunt).
Novisiblebruisesovertheabdomen.
Soonafterinjury.
Respiratorydistress.
Herniationofgutintolefthaemithorax.
USG Ba studies CT scan Operative Findings- - - Herniationof
smallgutthroughposterolateralrentin
diaphragm.Mode of Injury Duration* Clinical
FindingsPlain X-rays
Case11:MaleAge1
Roadsideaccident(blunt).
Bruisesoverleftlumbararea.
Soonafterinjury.
Respiratorydistress.
Herniationofgutintolefthaemithorax.
USG Ba studies CT scan Operative Findings- - - Herniationof
largegutthroughposterolateralrentin
diaphragm.Post-operativeperiodwasuneventfulforallcasesexceptCases9and11;bothpatientshadbronchopneumonia.*Durationbetweenoccurrenceoftraumaandhospitaladmission.Abbreviations:USG=ultrasonography,Ba=barium,CT=computerisedtomography.
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normally higher intrathoracic pressure andthe elasticity of the contracted diaphragm. Thediaphragmmost frequently tearsat the junctionof themuscular and tendinous elements that iscalledthecentrumtendinosum(5).Herniationofthestomach,smallandlargeintestines,kidneys,andspleenmayfollowtheruptureacutelyoryearsaftertheoriginalinjury.Delayedherniationoftheabdominal viscera may occur under a numberof circumstances. In some cases, thediaphragmweakenssecondarytotheaggressiveinflammatoryresponse at the ruptured site. Patients are alsoat risk just after tracheal extubation when theintrathoracicpressurequicklybecomesnegative.Alternatively, visceral content herniation mayoccur slowly, as the physiologically negativeintrathoracicpressuresgentlypulltheabdominalcontentsthroughthediaphragmaticdefect(6). Most diaphragmatic ruptures occur on theleftside.Thisisbelievedtobeduetoacongenitallyweaker left haemidiaphragm and the protectiveeffect of the liver on the right side (5). In ourstudy,allofthecaseswereleft-sided. While the classic physical signs ofdiaphragmatic herniation include unilateralbreath sounds, a scaphoid abdomen, and bowelsounds over the lung fields, these clues arenot consistently present. Many victims simplydemonstrate respiratory distress as their solepulmonaryfinding.Additionally,90% to95%ofindividuals with diaphragmatic ruptures haveother significant injuries. The most commonassociatedpathologies include lacerationsof thespleen, liver,andkidney;pelvic fractures;majorvesseldisruption; longbone fractures;andheadtrauma(7).Diaphragmaticinjuryfollowingblunttraumaremainsrareinchildrenandmaybemoredifficultto assess than in adults for both anatomicaland physiological reasons. The compliance ofthe paediatric chest wall may result in internalinjury in theabsenceof theexternalevidenceofmajor injury (8).Asminor injuries have causedruptures of the diaphragm, the timing of theimpact during the respiratory cycle is possiblymore important than the severity of the traumabecauseitcreatesasignificantpressuregradientacross thediaphragm.ChestX-ray is oneof themost important methods for the detection ofdiaphragmaticruptureandherniation.ThechestX-ray,however,isdiagnosticinonly25%to50%ofcases(9).SuggestivefindingsonthechestX–rayincludeaninterrupted,indistinct,orelevatedhaemidiaphragm, bowel loops or air-fluid levelsinthelungspace,andadisplacednasogastrictubeinto the chest. Rib fractures, pneumothoraces,
haemothoraces, lower lobe collapse, andpleuraleffusions are associated complications that areusually evident onplainfilms andmay increasethesuspicionofamoreextensiveinjury(9). Other imagingmodalities that are availabletotheemergencyphysiciantoassesstheintegrityofthediaphragmincludeultrasonography(USG)and CT scan (7). Diaphragmatic discontinuity,diaphragmatic thickening, segmentalnonrecognition of the diaphragm, intrathoracicherniation of the abdominal viscera, elevationof the diaphragm, and both haemothorax andhaemoperitoneumarestrongpredictorsofabluntdiaphragmaticrupture(10). As diaphragmatic tears do not closespontaneously,adiaphragmaticrupturerequiressurgical closure (5).Laparotomy is the favouredsurgical approach to acute diaphragmaticrupture,giventhatapproximately50%ofpatientswith blunt diaphragmatic injuries have otherintra-abdominalpathologies(9).Thoracotomyiscommonlyemployedtorepairchronicruptures. Successful surgical andbioprosthetic repairof TDR poses a serious challenge for surgeons.With the advent ofmolecular tools, proteomics,regenerative medicine and systems biology,surgeons, physicians, molecular biologists andbioengineers are now following a commontranslational path to reach a viable solutionfor successful tissue and organ reconstruction.To the best of our knowledge, there hasnot yetbeen a report of anymechanical strain-inducedexpressionofatendon-specificproteinthatleadstoavisiblephenotypiceffectintraumaticrupturethat contributes to traumatic diaphragmaticherniation. Neither is there any data availablethat substantiates gene or protein expression intraumatic diaphragmatic hernia or associatedmulti-organ complications. New strategiesranging from laparoscopic patch and intestinalsub-mucosa to stem cell and tissue-engineeredbioprosthetic construct-based strategies forthe repair and reconstruction of diaphragmatichernias have been attempted in severallaboratoriesaroundtheworld(11–15).However,most of the data for these types of studiescome from congenital cases, and these remainchallengingoptionsbecauseofhighcomplicationrates.Ongoingandfutureexperimentsinanimalmodelsfordevelopingfoetalcell-basedtherapieswill open newer avenues towards developing atranslational approach to successfully deal withtraumaticdiaphragmreconstructioninchildren.
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Conclusion
TDR, though uncommon, does occur andshouldremainadiagnosticpossibilityinchildrenbecause these patients do not undergo self-healing and require surgical correction of thedefect.Itshouldremainapossibilityevenincasesof remote trauma, as exemplified by one of ourpatientswhopresentedafteroneyearoftrauma.ACT scan is thebest imagingmodalitybecauseitclearlydepictstheanatomywith3-dimensionalreconstructions and highlights other associatedorgan injuries, which is extremely helpful intreating the patient. Stem cells and tissue-engineeredbioprosthetics,whicharebeingtestedin several laboratoriesaround theworld, canbemore relevant in such cases as they can betterintegrateintogrowingtissues.
Authors’ Contributions
Conceptionanddesign,statisticalexpertise:DBDObtainingoffunding,provisionofstudypatients:KNRCollectionandassemblyofdata:RNAnalysisandinterpretationofthedata,draftingofthearticle:SRCriticalrevisionofthearticle:SR,DBDFinalapprovalofthearticle:KNR,SR,DBDAdministrative, technical, or logistic support:KNR,SM
Correspondence
DrDharaBDhaulakhandiPhD Cell andMolecular Biology, Otolaryngology (AllIndiaInstituteofMedicalSciences)DepartmentofBiotechnology&MolecularMedicinePostGraduateInstituteofMedicalSciencesPtBDSharmaUniversityofHealthSciencesRohtak124001Haryana,IndiaTel:+91-9416312237Fax:+91-1262-211911Email:[email protected]
References
1. Rodriguez-Morales G, Rodriguez A, Shatney CH.Acute rupture of the diaphragm in blunt trauma:Analysisof60patients.J Trauma.1986;26(5):438–444.
2. Fataar S, Rad FF, Schulman A. Diagnosis ofdiaphragmatictears.Br J Radiol.1979;52:375–381.
3. Sharma LK, Kennedy RF, Heneghan WD. Ruptureof the diaphragm resulting from blunt trauma inchildren.Can J Surg.1977;20(6):553–556.
4. Shackleton KL, Steward ET, Taylor AJ. Traumaticdiaphragmatic injuries: Spectrum of radiographicfindings.Radiographics.1998;18(1):49–59.
5. Schumpelick V, Steinau G, Schluper I, Prescher A.Surgicalembryologyandanatomyof thediaphragmwith surgical applications. Surg Clin North Am.2000;80(1):213–239.
6. RamosC,KoplewitzB,BabynP,MansonPS,EinSH.Whathavewelearnedabouttraumaticdiaphragmaticherniasinchildren?J Pediatr Surg.2000;35(4):601–604.
7. Friedlaender E, Tsarouhas N. Traumaticdiaphragmatic rupture inapediatricpatient:Acasereport.Pediatr Emerg Care.2003;19(5):340–342.
8. Brandt ML, Luks FI, Spigland NA, DiLorenzo M,Laberge JM, Ouimet A. Diaphragmatic injury inchildren.J Trauma.1992;32(3):298–301.
9. RothrockS,GreenS,MorganR.Abdominaltraumaininfantsandchildren:Promptidentificationandearlymanagementofseriousandlife-threateninginjuries.Part II: Specific injuries and ED management.Paediatr Emerg Care.2000;16(3):189–195.
10. Nchimi A, Szapiro D, Ghaye B,Willems V, KhamisJ,HaquetL,etal.HelicalCTofbluntdiaphragmaticrupture.AJR Am J Roentgenol.2005;184(1):24–30.
11. Fuchs JR,KavianiA,Oh JT, LaVanD,UdagawaT,JenningsRW,etal.Diaphragmaticreconstructionwithautologous tendon engineered from mesenchymalamniocytes. J Pediatr Surg.2004;39(6):834–838.
12. Kunisaki SM, Fuchs JR, Kaviani A, Oh JT, LaVanDA,VacantiJP,etal.Diaphragmaticrepair throughfetal tissue engineering: A comparison betweenmesenchymal amniocyte- and myoblast-basedconstructs.J Pediatr Surg.2006;41(1):34–39.
13. FauzaDO,MarlerJJ,KokaR,R.ForseRA,MayerJE,Vacanti JP. Fetal tissue engineering: diaphragmaticreplacement.J Pediatr Surg2001;36(1):146–151.
14. HolcombGW3rd,OstlieDJ,MillerKA.LaparoscopicpatchrepairofdiaphragmaticherniaswithSurgisis.J Pediatr Surg.2005;40(8):E1–E5.
15. SandovalJA,LouD,EngumSA,FisherLM,BouchardCM,DavisMM,etal.Thewholetruth:Comparativeanalysisofdiaphragmaticherniarepairusing4-plyvs8-plysmallintestinalsubmucosainagrowinganimalmodel.J Pediatr Surg.2006;41(3):518–523.
