Introduction MirizzisyndromeMSisarelativelyrarecomplicationof gall-stonedisease.Nowadays,itisconsiderednotonlyasanextrinsic compressionofthecommonhepaticductbyastoneimpactedat thecysticd
Trang 1jo u r n al h om ep a g e :w w w e l s e v i e r c o m / l o c a t e / i j s c r
Acute acalculous cholecystitis complicated by MRCP-confirmed Mirizzi
syndrome: A case report
Yuri N Shiryajeva,b,∗, Anna V Glebovaa,b, Tatyana V Koryakinac, Nikolay Y Kokhanenkoa
a Department of Faculty Surgery named after Professor A.A Rusanov, Saint-Petersburg State Pediatric Medical Academy, Saint-Petersburg, Russian Federation
b Sixth Department of Surgery, Mariinsky Hospital, Saint-Petersburg, Russian Federation
c Department of MRI, Mariinsky Hospital, Saint-Petersburg, Russian Federation
a r t i c l e i n f o
Article history:
Received 30 October 2011
Received in revised form 5 November 2011
Accepted 15 November 2011
Available online 23 November 2011
Keywords:
Acute acalculous cholecystitis
Mirizzi syndrome
Magnetic resonance
cholangiopancreatography
a b s t r a c t
© 2011 Surgical Associates Ltd Published by Elsevier Ltd All rights reserved
1 Introduction
Mirizzisyndrome(MS)isarelativelyrarecomplicationof
gall-stonedisease.Nowadays,itisconsiderednotonlyasanextrinsic
compressionofthecommonhepaticductbyastoneimpactedat
thecysticductorgallbladderneck,butalsoaresultofa
cholecys-tobiliaryfistulacausedbyinflammationandpressurenecrosisof
thegallbladderandcommonhepaticductwallsbytheimpacted
stone.1–3 The most typical clinical symptoms in MS are upper
abdominal(especially rightupper quadrant)pain,jaundice and
fever.4–5SurgeryisthemosteffectivetreatmentofMS,butitis
accompanied withan increasedrisk of bile duct injury Severe
inflammationandadhesionsinthesubhepaticareaarevery
com-monandalmostalwaysinvolvethehepatoduodenalligamentand
Abbreviations: MS, Mirizzi syndrome; AAC, acute acalculous cholecystitis;
ERCP, endoscopic retrograde cholangiopancreatography; MRCP, magnetic
reso-nance cholangiopancreatography; US, ultrasonography.
∗ Corresponding author at: Department of Faculty Surgery named after Professor
A.A Rusanov, Saint-Petersburg State Pediatric Medical Academy, Litovskaya Str., 2,
194100 Saint-Petersburg, Russian Federation Tel.: +7 812 2757362;
fax: +7 812 2757326.
E-mail addresses: shiryajev@yandex.ru (Y.N Shiryajev), glebova.anna@mail.ru
(A.V Glebova), KoryaTV@yandex.ru (T.V Koryakina), kohanenko@list.ru
(N.Y Kokhanenko).
theydistortthenormalanatomicrelationshipsandproportions Therefore, it is highly desirable todiagnose MS preoperatively
to improve orientation in the surgical field and provide maxi-mal safety of the procedure The clinical symptoms of MS are non-specific, and are very similar to the symptoms of chole-docholithiasis.Ultrasonography(US)andcomputedtomography have limited sensitivity,3,6 and they are considered as screen-ingtests.AdiagnosisofMShastobeconfirmedbyothertests Themosthelpfuldiagnostictools toshow thevariationsinMS areeitherendoscopicretrogradecholangiopancreatography(ERCP)
orpercutaneoustranshepaticcholangiography.1,4However,these examinationsareinvasiveandmayleadtocomplicationsandeven fataloutcomes.Duringthelastdecademagneticresonance cholan-giopancreatography(MRCP)hasbeenwidelyusedanditisnow consideredasa goodnon-invasiveand averyaccurateimaging modalityinMSandinmanyotherdiseasesofthe hepatopancre-atobiliaryarea.7,8
Acuteacalculouscholecystitis(AAC)canleadtothe develop-mentofaconditionwhichisverysimilartoMSinitsclinicalcourse and imaging findings.The commonhepaticduct is compressed notbyastoneatthecysticductorHartmann’spouch,butbythe severelyinflamedandenlargedgallbladder.Suchcasesare exclu-sivelyrare.Wewereabletoidentifyonlythreewell-documented casesintheliterature.9–11Allthesepatientswereoperatedon,and
inallthreecasesapreoperativediagnosiswasmadebyERCP
2210-2612/$ – see front matter © 2011 Surgical Associates Ltd Published by Elsevier Ltd All rights reserved.
Trang 2Fig 1. MRCP demonstrated extrinsic compression of the common hepatic/common
bile duct by an inflamed, enlarged gallbladder without any stones in the biliary
system.
Recently,theauthorsobservedapatientwithjaundice,
devel-opedonthebackgroundofacutecholecystitis.OnUSexamination
stones in the gallbladder and bile ducts were not found The
signsofbiliaryobstructiondisappearedpromptlyonconservative
treatment.MRCPconfirmedsignificantcompressionofcommon
hepatic/commonbileductbytheinflamedandenlarged
gallblad-der,aswellasabsenceofstonesinthebiliarysystem.Conservative
treatmentwasundertaken,andsymptomsofacutecholecystitis
andjaundicegraduallyresolved
A52-yearmalepatientN.wasadmittedurgentlyon24.02.2011
withsevereupperabdominalpain,whichwasespeciallymarkedin
therighthypochondrium.Hehadnauseaandrepeatedbile
vom-iting.Thesecomplaintsstartedtwodaysagoandtheyincreased
withtime.Thiswashisfirstepisodeofsuchanattack.Pastmedical
historywasunremarkable.Physicalexaminationshowed
moder-atetendernessintherighthypochondriumandepigastrium.There
werenoperitonealsigns.Ortner’ssignandMurphy’s signwere
weaklypositive.Laboratorydatarevealedleucocytosis12,600/ml
USfindings:gallbladder103mm×38mm,anditswallwas
thick-enedto5mm.Thecommonbileductwasdilated(12mm);there
werenostonesinthegallbladderandinbileducts.After
crystal-loidinfusionwithdrotaverinandmetamizolesodiumthepatient’s
paindecreased,andvomitingstopped.Onthenextmorningthe
painincreasedagain,and hisskinandsclera becameyellowish
Palpationshowedseveretendernessandmildmuscularrigidityin
therighthypochondrium.Ortner’ssignandMurphy’ssignbecame
highlypositive.Biochemicalbloodtestsrevealedincreasein
biliru-bin(total 101mol/l, direct25mol/l)and transaminases(ALT
295IU/l(normalrange 0–55),AST123IU/l(5–34)), whereashis
amylaselevelremainednormal(46IU/l,normalrange25–110).A
repeatedUSshowedthegallbladdertobe110mm×43mm,itswall
wasnotthickened,intrahepaticbileductsandcommonbileduct
(3mm)werenotdilated.Gastroduodenoscopyshowedno
signifi-cantchanges
AAC,complicatedwithjaundice,wasdiagnosedonthebasisof
clinical,laboratoryandinstrumentalfindings.Conservative
man-agementwascontinuedwithadditionofantibiotics.MRCPonthe
Fig 2.Check MRCP 5 months later demonstrated post-inflammatory changes of the gallbladder which became reduced in size and was deformed However its wall was not thickened The intra- and extrahepatic bile ducts were not dilated and free of stones.
3rddayafteradmissiondemonstrated enlargementofthe gall-bladderwithitswallthickeningandextrinsiccompressionofthe commonbileductwithoutanystonesinthebiliarysystem(Fig.1) Thepatient’spainandjaundicegraduallyresolvedwith conserva-tivetreatment,andlaboratoryfindingssignificantlyimprovedto normal/subnormalranges.Hewasdischargedontheninthday
Oncheck-upintheclinic5monthslaterthepatient demon-strated no complaints, no jaundice,his abdomen wassoft and notpainful.USfindingswereunremarkable.ThefollowupMRCP (Fig.2)showedthegallbladdertobereducedinsizeandit was deformed.Itswallwasnotthickened(3mm)andwithoutany fill-ingdefects.Theintra-andextrahepaticbileductswerenotdilated andfreeofstones
3 Discussion
AAC can be complicated with extrinsic compression of the commonhepatic/commonbileductbytheenlargedandinflamed gallbladder,whichwasfollowedbyjaundice.Itsmechanismisvery similartoMS,whenthebileductiscompressedfromoutsidedue
toastoneimpactedatthegallbladderneckorcysticduct.Thefirst descriptionofthisconditionusingUSandERCPwaspublishedby R.J.Ippolitoin1993.9 Averysimilarcasewasdescribed by Ital-ianauthorsin1992.12Theydescribedayoungmalepatientwho complained of repeatedattacksof right upperabdominal pain, accompaniedwithjaundice,though,thebiliaryobstructionwas notcausedbyAAC,butbyanextremelyenlarged“congestive” gall-bladderdueto“cysticductsyndrome”.Compressionofthecommon hepatic/commonbileductwasconfirmedbyERCPand intraopera-tively.Intheliteraturewemanagedtofindonlytwoadditionalcase reportsofAAC,complicatedwiththebileductcompressionand jaundice.10,11Thus,thispathologicalentityisexceptionallyrare
Itisinterestingtonotethat theterminologytodescribethis conditionhasnotbeenagreedupon.Theauthorsofthesethree publishedcasereports useddifferentterms.InIppolito’spaper9
hecalledthecondition“Acuteacalculouscholecystitisassociated withcommonhepaticductobstruction:avariantofMirizzi’s syn-drome”.K.Mergeneretal.,10entitledtheirarticle“Pseudo-Mirizzi syndromeinacutecholecystitis”,whileS.Ahlawat11usedthetitle –“AcuteacalculouscholecystitissimulatingMirizzisyndrome:a
Trang 3canbeconsideredtobeaspecialkindofMS
OneMS classificationproposedbyJapanese authorsin1997
basedontheirexperiencein30cases.5TheydefinedtypeIVofMS
as“hepaticductstenosisasacomplicationofcholecystitisinthe
absenceofcalculiimpactedinthecysticductortheneckofthe
gall-bladder”.Therewere8suchcasesintheauthors’series.However,
neitherthefigure,northepresentationofatypicalcaseallowed
thereaderstosaywhethersomeofthese8patientshadacute
acal-culouscholecystitisornot.WeconsiderthatthecasesofMSwith
acalculouscholecystitiscanbeclassifiedasNagakawa’stypeIV
T.Nagakawaetal.believe,that“forTypeIVdisease,whichoften
presentswithseverestenosisofthehepaticduct,reconstructionof
thebiliarytractfollowingexcisionofthestrictureis indicated”
However,wethink, sucha procedureistoomajorand
unjusti-fied.Inflammatoryhepaticocholedochealstrictureshealwellafter
simplecholecystectomy,whencombinedwithbiliarydrainage(by
transhepaticornasobiliarydrainageasperformedinthefirststage
ofsurgicaltreatment,orbyoperativeT-tubedrainage).R ˇColovi ´c
etal.proposedtheirownclassificationforMS.13Theseauthors’MS
typeIVisthesameasNagakawa’sclassification.However,their
conceptionofsurgeryissignificantlydifferent,5anditisvery
sim-ilartoourapproach
The diagnosis of MS in acalculous cholecystitis is usually
suggestedbyUSandERCP,9–12orsometimesbypercutaneous
tran-shepaticcholangiography.12Asthefirststageofsurgicaltreatment,
choledochalstentingwasused10torelievejaundice.Allthepatients
wereoperatedon:inthreecasesopencholecystectomy,9,11,12 in
one laparoscopic cholecystectomy10 was performed In all the
three cases of AACthere was gangrenousinflammation of the
gallbladder.9–11 Despite the absence ofbiliary drainage (itwas
placedonlyin1of4patients),jaundiceandothersymptoms
suc-cessfullyresolved
4 Conclusion
Ourcase canbeconsideredasextraordinarybecauseof two
factors.First,thediagnosisofMSinacalculouscholecystitiswas
confirmedbyMRCP,whichappearstobethefirsttimereported
intheliterature.Second,conservativetreatmentwaseffectiveand
surgerywasavoided.ItallowedustoperformacheckMRCP,which
showedresolutionofacuteinflammationofthegallbladder,aswell
asdecompressionofthecommonhepatic/commonbileduct
Conflict of interest
None
Funding
None
Ethical approval
Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereport.Acopyofthewrittenconsentis avail-ableforreviewbytheEditor-in-Chiefofthisjournalonrequest
Author’s contributions
YuriN Shiryajev:ideaof thearticle,study design,data col-lection, data analysis,literature search, writing, revision of the manuscript
AnnaV.Glebova:datacollection,dataanalysis,literaturesearch TatyanaV.Koryakina:datacollection,dataanalysis,makingand interpretationofultrasoundandMRIexamination
NikolayY.Kokhanenko:criticalrevisionandsupervisionofthe work
Allauthorsreadandapprovethefinalversionofthemanuscript
Acknowledgements
None
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