Minimally Invasive Repair of Morgagni Hernia - A Multicenter Case Series

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Please cite this article in press as: Lamas-Pinheiro R, et al. Rev Port Pneumol. 2016. http://dx.doi.org/10.1016/j.rppnen.2016.03.008

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RevPortPneumol.2016;xxx(xx):xxx---xxx

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ORIGINAL

ARTICLE

Minimally

invasive

repair

of

Morgagni

hernia

---

A

multicenter

case

series

R. Lamas-Pinheiro

a,∗

,

J. Pereira

b

,

F. Carvalho

b

,

P. Horta

c

,

A. Ochoa

c

,

M. Knoblich

d

,

J. Henriques

d

_,

_T.

_{Henriques-Coelho}

a

_,

_J.

_{Correia-Pinto}

a

_,

_P.

_Casella

d

_,

_J.

_{Estevão-Costa}

a a_Pediatric_Surgery_Department,_Faculty_of_Medicine,_Hospital_São_João,_Porto,_Portugal

b_Pediatric_Surgery_Department,_Centro_Hospitalar_do_Porto,_Porto,_Portugal c_Pediatric_Surgery_Department,_Hospital_Pediátrico_de_Coimbra,_Coimbra,_Portugal

d_Pediatric_Surgery_Department,_Centro_Hospitalar_Lisboa_Central_Hospital_D._Estefânia,_Lisboa,_Portugal

Received30November2015;accepted6March2016

KEYWORDS Laparoscopy; Minimallyinvasive surgery; Percutaneous; Morgagnihernia; Diaphragmatichernia

Abstract Childrenmaybenefitfromminimallyinvasivesurgery(MIS)inthecorrectionof Mor-gagnihernia(MH).ThepresentstudyaimstoevaluatetheoutcomeofMISthroughamulticenter study.

NationalinstitutionsthatuseMISinthetreatmentofMHwereincluded.Demographic,clinical andoperativedatawereanalyzed.

ThirteenpatientswithMH(6males)wereoperatedusingsimilarMIStechnique(percutaneous stitches)atameanageof22.2±18.3months.Sixpatientshadchromosomopathies(46%),five withDownsyndrome(39%).Respiratorycomplaintswerethemostcommonpresentation(54%). Surgery lasted 95±23min. In noneofthepatients was the herniasac removed; prosthesis was neverused.In theimmediatepost-operativeperiod,4patients(36%)were admittedto intensivecareunit(allwithDownsyndrome);allpatientsstartedenteralfeedswithinthefirst 24h.Withameanfollow-upof56±16.6months,thereweretworecurrences(18%)atthesame institution,oneofwhichwasrepairedwithanabsorbablesuture;bothwithDownsyndrome.

TheapplicationofMISintheMHrepairiseffectiveeveninthepresenceofcomorbiditiessuch asDownsyndrome;thelatterinfluencestheimmediatepostoperativerecoveryandpossiblythe recurrencerate.Removalofherniasacdoesnotseemnecessary.Non-absorbablesuturesmay bemoreappropriate.

is an open access article under the CC BY-NC-ND license (http://creativecommons.org/

licenses/by-nc-nd/4.0/).

∗_{Corresponding}_author.

E-mailaddress:rubenlms@hotmail.com(R.Lamas-Pinheiro). http://dx.doi.org/10.1016/j.rppnen.2016.03.008

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Introduction

The Morgagni foramen was first described by the Italian anatomistGiovanni Morgagniin1769.1 _This_defect_located

intheanteromedialportionofthediaphragm,betweenthe xiphisternal and costal margin fibers, results from a fail-ureinthefusionoftheseptumtransversumandthecostal arches.2 _Its _presence _may _allow _the _passage _of

abdomi-nalcontents,includingtheomentum,stomach,colon,liver or small bowel into the thoracic cavity, thus resulting in Morgagni hernia (MH).3,4 _MH _is _extremely _rare, _occurring

approximatelyin1outof4800livebirths,andaccountsfor lessthan6%ofallcongenitaldiaphragmaticdefectsrepaired inpediatricagegroup.5_Associated_congenital_malformation

such ascongenital heart disease, chest wall deformities, intestinalmalrotation or omphalocele hasbeen reported. Chromosomalanomalies, mainlyDownsyndrome (DS),are frequentfindings.2,6_MH_is_typically_asymptomatic_and_may

present withnonspecific respiratory symptoms, often dis-coveredlaterinlife.2,7

Thepresentstudyaimstoassessandevaluatethesurgical andclinicaloutcomesinthemid-termfollow-upofchildren submitted to MIS in the correction of the MH, through a nationalmulticentricstudy.

Material

and

methods

All national institutions that use MIS in the treatment of MHinchildrenwereincludedinaretrospectivetransversal

study. Clinical files from all patients operated on from December2006toJune2013werereviewed andanalyzed by members of each institution. Patients were included whensubmittedtolaparoscopic-assistedsurgicalapproach usingthreeportsandwhen sutureswere performedwith, separated, percutaneous, ‘‘U’’ shaped, stitches, through the full thicknessof the anteriorabdominal wall (Fig. 1) and the knots were tied in the subcutaneous tissue, by separated minor skin incisions or by a single incision. Patientssubmittedtolaparotomy,thoracotomyoranyother minimal invasive technique different than that described were excluded. Collected data included age, gender, clinical presentation, associated malformations, surgical details, length of hospital stay, post-operative admission to intensive care unit, time of start of enteral feeds, complicationsandfollow-updetails(clinical,radiological).

Results

Duringthe studyperiod,13 patients (sixmalesand seven females)werediagnosedwithMHandsubmittedto correc-tionatfourdifferenttertiarycenters.Demographicdatais presented inTable1.Sixpatientshadchromosomopathies (46%) including five children with Down syndrome (39%). The mostcommonpresentation wasrespiratorysymptoms (54%), followed by incidental finding(23%). Five patients (39%)hadpreviousadmissionsfordifferentcauses. Diagno-siswasreachedbychestroentgenogram,contrastenemaor computedtomographicscan.

Figure1 Operativeviewofthedefect(A);stitchesincludingplicationofthesac(B)anddiaphragmaticrim(C);finalaspectafter knotstying(D).

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Table1 Demographicsandclinicalpresentation.CXR---plainchestroentgenogram;CT---computedtomography.

Case Gender Clinicalpresentation Previous

admissions Ageatdiagnosis (months) Chromosomopathy Diagnosis 1 F Pneumonia Yes 7 No CT 2 F Persistentcoughing No 5 No CXR

3 M Bronchiolitis Yes 4 Downsyndrome CXR,CT

4 F Bronchiolitis No 14 No CXR,CT

5 M Fever No 9 No CXR,CT

6 F Incidentalduringechocardiogram Yes 55 Downsyndrome CXR,CT

7 M Incidentalduringechocardiogram Yes 17 Downsyndrome CXRCT

8 M Incidental,afterforeignbody

ingestion

Yes 48 Downsyndrome CXR,CT

9 F Nonspecificrespiratorysymptoms --- 22 46,XX,del(4) CXR

10 M Bronchiolitis Yes 18 No CXR,CT

11 F Asymmetricchestwalldeformity No 16 No CXR,CTcontrast

enema

12 F --- --- 19 No Rx

13 M Bronchiolitis Yes 14 Downsyndrome CXR,contrast

enema

Inallcases,thecorrectionwasinitiallyperformedusing identical laparoscopic-assisted technique with full thick-nessseparatedpercutaneous stitches.Surgical details are expressedinTable2.Childrenwereoperatedonatamean age of 22±18.3 months. The mean operative time was 95±23min. There were no intra-operative complications andnoneedforconversion.Innoneofthepatientswasthe herniasacremoved,andprostheticpatchwasneverused.In theimmediatepost-operativeperiod,4patients(36%)were admittedtointensivecareunit,allofthemwithDown syn-drome(p<0.05).Allpatientsstartedenteralfeedingwithin thefirst24h.

Withameanfollow-upof56±16.6months,therewere tworecurrences(18%)at thesameinstitution.Oneof the recurrenceswasacase inwhichanabsorbablesuturewas usedandbothhadDownsyndrome,butthesefindingswere not significantly associated with recurrence (p=0.15 and

p=0.13respectively)

Discussion

Surgical repair of MH, with reduction of hernia contents and primary closure of the defect, is generally indicated inordertopreventpotentialmajorintestinalcomplications suchas,obstruction,volvulusorperforation.Althoughsome authors advocatetransthoracic surgery arguing for a bet-terexposureandbettervisualizationof thephrenicnerve andsafersacresection,themajorityofpediatricsurgeons prefer transabdominal approach. For many years, thora-cotomy and specially laparotomy have been the standard surgical approaches. After the firstlaparoscopic repair of MH by Kuster et al.20 _in _1992, _minimally _invasive

tech-niques,includingsingle-portapproachandroboticsurgery, becamerapidlyacceptedaselectedapproachesintherepair of MH in both childrenand adults.7,16,21---23 _Many_technical

aspectsarestillunderdebate.The laparoscopicreduction of the hernia contentsdoes notappear tobe a problem,

but the method for closure of the defect is variableand maybetechnicallydemandingwhenperformedexclusively bylaparoscopy.Someauthorsrecommendthe laparoscopic-assistedrepairofMorgagniherniawithextracorporealknots underthesubcutaneoustissue.5,8---10_It_includes_the_full

thick-nessof the anterior abdominal wall in U-shaped stitches underdirectvision,sothatthesuturebecomessolid, allow-ingformaximumstrengthrepair.Bycontrast,anchoringthe suturesinthebackofthesternumandcostalmarginis tech-nicallychallengingandthefasciamaynotbestrongenough. Regardingthechoiceofsutureline,asourseries corrobo-rate,theuseofabsorbablesuturemaybeassociatedwith recurrence.24---26

Anothercontroversial issue iswhether or nottoexcise the hernia sac. Excision is suggested in order to reduce recurrence rate, but it may be laborious and potentially dangerous.10,12---15_As_advocated_by_others,_in_our_series_the

sacwasonlyplicated, thusavoidinganypossible injuryof the pericardium, pleura, or phrenic nerve that might be associatedwiththehernia sacexcision. Simultaneously,a lowrecurrenceratewasobserved.

TheliteraturerecordsofseriesofMHsubmittedto min-imalinvasiverepairarescarce withsmallsamplesizeand short-termfollow-up.Thecurrentmulticenterseriesisone ofthe largestwith thelongest reportedfollow-up (25---78 months)andwe havefounda15% (2/13)recurrencerate. Theremaining85%ofpatientsshowcomplete obliteration oftheresidualcavityinchestroentgenogram,aspreviously described.27,28

InlargeBochdalekcongenitaldiaphragmatichernias,the use of a prosthetic patch is frequently advocated. In MH repair,theuseofpatchismorecontroversial.Inaprevious series,ithasbeenhypothesizedthattensionclosurecould bethe cause of a high recurrence rate and the use of a patchwas suggested in orderto reduce tension of repair anddecreasedrecurrencerate.11,26,29_Our_series_have

con-tradictingresultsaswe reportaserieswithout theuseof patchandwithalowrecurrencerate.

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Please cite this article in press as: Lamas-P inheiro R, et al. Re v P ort Pneumol. 2016. http://dx.doi.org/10.1016/j.rppnen.2016.03.008

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Table2 Perioperativeandfollow-updetails.CXR---plainchestroentgenogram;ICU---intensivecareunit.

Case Ageat surgery (months) Laterality Hernia contents Suture Associated procedures Operative time(min) Needfor ICU Lengthof hospital stay(days) Residual cavity (CXR) Recurrence Follow-up (months)

1 8 Left Smallbowel Nonabsorbable No 60 No 1 No No 62

2 6 Bilateral Liver,Small

bowel

Nonabsorbable No 95 No 2 No No 42

3 4 Bilateral Colon Nonabsorbable Inguinalhernia

repair 105 Yes 16 No No 36 4 16 Right Small bowel, Colon Absorbable No 90 No 3 No No 49 5 9 Bilateral --- Nonabsorbable No 40 No 1 No No 60

6 61 Bilateral Liver,Colon,

Omentum

Nonabsorbable No 110 Yes 3 --- Yes 34

7 17 Bilateral Colon Absorbable Orchidopexy 115 Yes --- --- Yes 30

8 58 Bilateral Colon Nonabsorbable Adenoidectomy

Myringotomy

120 Yes 7 No No 59

9 23 Bilateral --- Nonabsorbable No 120 No 2 No No 46

10 23 Bilateral Smallbowel Nonabsorbable No 90 No 3 No No 24

11 17 Bilateral Colon Nonabsorbable No 105 No 3 No No 17

12 33 --- --- --- --- 105 No 3 No No 51

13 14 Bilateral Liver Nonabsorbable Orchidopexy

Myringotomy

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Theassociationwithchromosomaldisordersand congen-italanomaliessuchascongenitalheartdisease,chestwall deformities, intestinal malrotation, omphalocele, trisomy 21, pentalogyof Cantrell, Noonansyndrome,Prader---Willi syndrome and Turner syndrome is well established.2,6 _In

ourseries,six patients (46%)hadchromosomopathies and another one presented a chest wall deformity. As in ear-lier reports, Down syndrome was a frequent association (38%)18,19_; _all _the _patients _in _whom _recurrence _occurred,

alsohadDownsyndrome.Thischromosomopathyhasbeen previously associated with impaired healing, and there might be an association withrecurrence, although it was notstatisticallysignificantinourstudy.Additionally admis-sionstointensivecareunitsweresignificantlymorefrequent in patientswiththissyndrome,probablydue toother co-morbidities.18,19

In conclusion, laparoscopic-assisted repair of Morgagni herniausingsuturesincludingthefullthicknessofanterior abdominalwallandextracorporealknotsprovedtobe effec-tive,safe,andreliableinchildren,eveninthepresenceof comorbiditiessuchasDownsyndrome;thelatterseemsto influencetheimmediatepostoperativerecoveryand possi-blytherecurrencerate.Ourseriescorroboratesthatthere isnoneed forherniasac excisionor theuseofprosthetic patch; absorbablesutures arenot recommended. Further prospective randomized trials are needed to confirm and comparethesefavorableoutcomesoflaparoscopic-assisted repairofMorgagnihernia.

Ethical

disclosures

Protection of human and animal subjects.The authors declarethatnoexperimentswereperformedonhumansor animalsforthisstudy.

Confidentialityofdata.Theauthorsdeclarethatnopatient dataappearinthisarticle.

Right to privacy and informed consent.The authors declarethatnopatientdataappearinthisarticle.

Conflicts

of

interest

Theauthorshavenoconflictsofinteresttodeclare.

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