Clinical Medical & Case Reports
Open Journal of
ISSN2379-1039
Volume2(2016)Issue24
HochwaldS
OpenJClinMedCaseRep:Volume2(2016)
Gastritiscysticaprofunda:AchallengingdiseasediagnosedusinganovelapproachDanishShahab,MD;EmmanuelGabriel,MD;MoshimKukar,MD;AndrewBain,MD;StevenHochwald,MD*
*StevenHochwald,MD
DepartmentofSurgicalOncology,RoswellParkCancerInstitute,Buffalo,NY14263USA
Tel:(716)845-8244.
Abstract
Gastritiscysticaprofunda(GCP)isararehyperplasticlesionofunclearetiology.Clinicalsymptomsof
GCParevariableandrangefromnonspeci�icabdominalpaintogastricoutletobstruction.Oftenthe
diagnosisofGCPhasbeendif�iculttomakeassimpleesophagogastroduodenoscopy(EGD)guidedbiopsy
canfailtoyieldthediagnosis.Here,wereportacaseofGCPthatpresentedinachallenginganatomic
location,whichrequiredalaparoscopicintragastricsurgicaltechniquetomakethediagnosis.
Introduction
Gastritiscysticaprofunda(GCP)isarare,benigndiseasecharacterizedbypolypoidhyperplasia
andcysticdilatationofthegastricglandsthatextendintothesubmucosaofthestomach.Firstdescribed
in1947byScottandPayne,itwasn'tuntil1972thatLittlerandGilbermannsuggestedthatthepresence
ofcysticallydilatedgastricglandsinthesubmucosawasareactive,postsurgicalconditionforwhichthey
coinedtheterm“gastritiscysticapolyposa”[1].Thiswouldeventuallychangetothenowpreferredterm
“gastritiscysticaprofunda”becauseitresembledthesimilarlynamedconditioninthecolon[1].Clinically,
patientscanpresentwithupperabdominalpain,acidre�lux,nausea,anorexia,orbleeding;although
somepatientsmayexperiencenosymptoms[2].Inseverecases,patientscanexperiencemassiveupper
gastrointestinalhemorrhageandgastricoutletobstruction[2].GCPisoftenseenasgiantgastricfolds,
submucosal tumors,or isolatedpolyps [2].The lesionhasbeendescribedprimarily in theoperated
stomach, leading to thehypothesisofpriorgastricwall injuryas thepredominantcauseofGCP, [2]
although ithasbeendescribed in theunoperatedstomachaswell.Although theexactpathogenesis
mechanismremainslargelyunknown,GCPhasbeenfoundtobeassociatedwithischemiaandchronic
in�lammation[3].Itisalsothoughttobeapossibleprecancerouslesionsinceafewearlygastriccancers
havebeenassociatedwithit[4].Interestingly,thereisalsoanincreasedassociationincancerswithGCP
toEpstein-Barrvirus(EBV)positivepatients[5]andMenetrierdisease(MD)[6].Here,wedescribeacase
ofadif�iculttodiagnosegastricmassthatwasfoundtobeGCP.
CasePresentation
Ourpatientwasa72yearoldmalewithapastmedicalhistoryofanon-smallcelllungcancer
Keywordsgastritiscysticaprofunda;gastricglands;submucosaltumors
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treated with chemotherapy and radiation, coronary artery disease, and cardiomyopathy. Previous
esophagogastroduodenoscopy(EGD)performedatanoutsidefacilityinFebruaryof2014forsymptoms
ofre�luxshowedcircumferentialBarrett'sesophagusandasmalltomoderatehiatalhernia.Atthistime,
hewasreferredtoourinstitutionforcontinuedsurveillanceoftheBarrett'sesophagus.
InOctoberof2015,asurveillanceEGDshowedanewmassinthegastriccardiameasuring2.6x1.4
cm.Fineneedleaspiration(FNA)performedatthistimewasnondiagnostic.Themasswaslocatednear
the hiatal hernia pinch and was thought to represent a gastrointestinal stromal tumor (GIST) or
leiomyoma.Approximately6monthslater,anintervalEGDalsoanincreaseinthesizeofthegastriccardia
mass to approximately 4 cm (Figure 1). On endoscopic ultrasound (EUS), a heterogeneous mass
measuring 3.9 by 3.1 cm in the gastric cardia with small internal cystic spaces was described. He
underwent a second FNA of themass,which showed only in�lammation and again failed to yield a
diagnosis.ACTscanshowedasmoothwellmarginatedmassintheregionofthegastriccardiaandhiatal
hernia,suggestiveofasubmucosallocation(Figure2).
Giventheconcernforneoplasmbasedonitsincreaseinsize,surgicalresectionwasrecommended.
GiventhechallenginglocationofthelesionclosetotheGEjunction,alaparoscopicintragastricapproach
withendoscopicassistancewasrecommended.Thisisanovelapproachwherebythelaparoscopicports
areinsertedthroughtheabdominalwallandthenintothestomachunderdirectendoscopicvisualization.
Duringtheprocedureuponmanipulationofthemass,purulentdrainagewasnotedfromtheareaofthe
mass,whichresultedincollapseofthemass.Sincethemassdecompressed,wedecidednottoresectit.
Therewasnoevidenceofmalignancyonpreviousbiopsies.Thus, the justi�icationforamoreradical
operationwasnotpresent.However,priortocompletionofsurgerymultiplecoreneedlebiopsiesofthe
masswereobtainedusingthelaparoscopicintragastricapproach.Thepatienttoleratedtheprocedure
well.Hewasdischargedonpostoperativeday3withoutcomplications.Surgicalpathologyshowedthat
themasswasconsistentwithgastritiscysticaprofunda(Figure3).
Discussion
Gastritiscysticaprofundaisaconditioncharacterizedbybenign,cysticgrowthofgastricglands
into the submucosa of the stomach [7]. Although GCPs are thought to be associatedwith previous
gastrectomy,thislesioncanalsooccurwithoutpreviousgastricoperations,[8]asobservedinourcase.
GCPisoftenlocatedintheposteriororanteriorwallofthegastricbodyandintheintermediatezone
betweenthefundicandpyloricglands[9].Ingeneral,laboratorytestsarenotusefulinmakingadiagnosis
ofGCP[3].ThepathogenesisofGCPislikelyduetochronicischemiaandin�lammation.Thedisruptionof
integrity ofmuscularismucosa causes themigration of epithelial content into the submucosawith
subsequentatrophicgastritis,intestinalmetaplasiaandcysticdilatationofgastricglands[7].Gastritis
cysticaprofunda isabenign lesion, althoughapossibleprecancerousnaturehasbeenhypothesized
[10].GCP has been shown to occur more frequently in the presence of gastric cancers [11]. In a
pathologicalstudyof10,728patientswithgastriccancer,GCPwasfoundin161patients[11].
ThereappearstobeapathogeneticroleofEpstein-Barrvirus(EBV)inthedevelopmentofGCPand
cancer[8].IthasbeendemonstratedthatthereisadelayofapoptosisinEBV-positivegastriccarcinomas
associatedwithupregulationofBCL-2andp53,andadecreaseincellulardifferentiationassociatedwith
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a decrease in E-cadherin expression [8]. EBV infection can alter the normal cell cycle, resulting in
disruptioninthecellularprocessesandcheckpointsresponsibleforregulatingcelldivision,whichthen
resultsinthecarcinogeneticeffectsofEBV[8].EBVtitershavebeenshowntobesigni�icantlyhigherin
dysplasticgastricmucosaversusnon-neoplasticgastricmucosa.InastudydonebyKimetal,theauthors
showed thatwithin the transitional areabetweenGCP and gastric carcinoma, thereweredysplastic
changespositiveforEBV[8].AnothergroupfoundthattheEBVpositiveratewassigni�icantlyhigherina
GCPgastriccancergroup(31.1%)thaninanon-GCPgastriccancergroup(5.8%),whichsuggeststhatGCP
wassigni�icantlyassociatedwithEBV-positivegastriccancersandthatEBVinfectionsmayplayarolein
thedysplasticchangesassociatedwithGCP[11].
Menetrierdisease(MD)isanotherpremalignantconditionwhichhasshownsomeassociation
withGCP[12].MDisanuncommon,idiopathic,hyperplasticgastropathycharacterizedbyhyperplasiaof
foveolarmucouscells,whichresultsinthickeningofgastricfoldsandhypoalbuminemia[6].Itmostly
involvesthegastricfundusandbody[6].Therehavebeenmanyreportsshowingcloseassociationwith
MDandGCPaspotentialprecancerouslesions[6].Furtherwork-upincludestestingforcytomegalovirus
and H. pylori. Treatment options include proton pump inhibitors (PPI) or H2 blockers, octreotide,
monoclonalantibodiestoepidermalgrowthfactorreceptor(EGFR),andgastrectomy[6].
RegardingthediagnosisofGCP,standarddiagnosisbyEGDisoftendif�icultbecausethestandard
FNAbiopsyspecimenisusuallylimitedtothemucosa,whichcannotprovidesuf�icientinformationabout
thedeepersubmucosa[13].Infact,inmanycasesthepreoperativediagnosisofGCPremainschallenging
despitethecurrentadvancesinendoscopictechniquesandthuspatientsmayhavetoundergosurgical
resectionfor�inalpathologicdiagnosistoguidetreatment[13].Oftentimesandinourcase,CTscanand
EUShavebeenusedasacomplementarytooltodelineateadditionalcharacteristicsoftheselesions[13].
ThemostcommonendoscopicfeaturesofGCPbyconventionalwhite-lightendoscopyarenonspeci�ic[3].
Infact,manygastroenterologistsnowsuggestthatthediagnosticmodalityofchoiceisEUS[3].GCPon
EUSshowsprimarily3majorechoicpatterns:anechoic(35.3%),mixedheterogeneouswiththickened
overlyingmucosa(50%),andhypoechoicwithmicrocysts(14.7%)[3].The�inaldiagnosishoweverstill
hastobedeterminedbyhistologicalexam[3].
ThereiscurrentlynoconsensusontheoptimalGCPtreatment.Duetoaninsuf�icientamountof
informationonGCP,therehavebeenavarietyoftreatmentrecommendations,rangingfromobservation
toradicalresection[3].XuGetaldevelopedastandardprotocoltosystemicallyinvestigatetheselesions
withEUSbeforeendoscopicsubmucosalresection/endoscopicsubmucosaldissection(EMR/ESD)[3].
GiventhefactthatthediagnosisofGCPmainlyreliesonhistopathology,endoscopicresectionservesboth
adiagnosticandtherapeuticprocedure.Ifperformedsuccessfully,endoscopicresectionwithEMRorESD
islessinvasive,safer,andmoreeconomicalthanopensurgicalmethods[3].Moreimportantly,endoscopic
resectionofGCPbetterpreservesgastricfunctionwithminimalinjury[3].Itshouldbenotedtherewere
limitations to theirproposedprotocol includingsmall samplesize, single institutionexperience,and
retrospectivenatureofthestudy.
Inourcasereport,wepresentedapatientwhoselesionwasfoundtobeinachallengingproximal
anatomiclocationassociatedwithahiatalhernia.ItwasincloseproximitytotheGEJ,thusmakingit
unresectablethroughastandardlaparoscopicapproach.Thus,alaparoscopicintragastricapproachwith
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endoscopicassistancewasattempted.However,themassdecompressedandwasnolongeramenableto
resection. Two previous endoscopic-guided FNAs failed to establish the diagnosis. Therefore, we
performed core biopsies through the intragastric port, which successfullymade the diagnosis. This
approachwasfoundtobediagnosticallyeffectiveandhadtheadvantageofstayingminimallyinvasive.In
conclusion,GCP isa rareandoftendif�icultdiagnosis tomake,but incertaincasessuchasours the
diagnosis can be facilitated using a novel surgical method consisting of a laparoscopic intragastric
approachwithendoscopicassistance.
Figures
Figure1:(A)EGDshoweda4cmsubepithelialmassinthegastriccardiaatthehiatalherniapinchlocatedat40cm.
(B)OnlimitedEUS,therewastheheterogeneousmassmeasuring39mmby31mminthegastriccardiawithsmall
internalcysticspaces.
Figure2:(A)ThepatientCTscanshoweda3.5x1.7cmsmoothlymarginatedmassadjacenttothegastricwalljust
beyondtheGEjunctioninthegastriccardia(arrow).(B)Therewasalsoahiatalhernia(arrowhead).Differential
diagnosisincludedgastrointestinalstromaltumor(GIST)andleiomyoma.
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Figure3:(A)Lowpower40xand(B)Highpower100xmagni�ication.Thelaparoscopicintragastriccoreneedle
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ManuscriptInformation:Received:November10,2016;Accepted:December27,2016;Published:December29,2016
1 2 2 3AuthorsInformation:DanishShahab,MD ;EmmanuelGabrielMD ;MoshimKukarMD ;AndrewBainMD ;StevenHochwald2*MD
1DepartmentofMedicine,UniversityatBuffalo,Buffalo,NY14263USA2DepartmentofSurgicalOncology,RoswellParkCancerInstitute,Buffalo,NY14263USA3DepartmentofMedicine,RoswellParkCancerInstitute,Buffalo,NY14263USA
Citation:ShahabD,GabrielE,KukarM,Bain,Hochwald.Gastritiscysticaprofunda:achallengingdiseasediagnosedusinga
novelapproach.OpenJClinMedCaseRep.2016;1204
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