www.elsevier.pt/ge
CLINICAL
CASE
Groove
Pancreatitis
with
Biliary
and
Duodenal
Stricture:
An
Unusual
Cause
of
Obstructive
Jaundice
Marta
Gravito-Soares
a,∗,
Elisa
Gravito-Soares
a,
Ana
Alves
a,
Dário
Gomes
a,
Nuno
Almeida
a,
Guilherme
Tralhão
b,
Carlos
Sofia
aaGastroenterologyDepartment,CentroHospitalareUniversitáriodeCoimbra,Coimbra,Portugal bSurgeryDepartment,CentroHospitalareUniversitáriodeCoimbra,Coimbra,Portugal
Received2June2015;accepted6September2015
KEYWORDS Cholestasis; Duodenal Obstruction; ObstructiveJaundice; Pancreatitis Abstract
Introduction:Groovepancreatitisisanuncommoncauseofchronicpancreatitisthataffects thegrooveanatomicalareabetweentheheadofthepancreas,duodenum,andcommonbile duct.
Clinicalcase: A 67-year-old man with frequent biliary colicand analcohol consumption of 30---40g/daywasadmittedtothehospitalcomplainingofjaundiceandpruritus.Laboratory anal-ysisrevealedcholestasisandtheultrasoundscanshowedintra-hepaticbiliaryductsdilatation, middlethirdcysticdilatationofcommonbileduct,enlargedWirsungandpancreaticatrophy. Themagnetic resonancecholangiopancreatographyshowed imagingfindingscompatiblewith groove pancreatitis.An esophagogastroduodenoscopylaterexcluded duodenalneoplasia.He wassubmitted toaRoux-en-Ycholangiojejunostomybecauseofcommonbileductstricture. Fivemonthslateragastrojejunostomywasperformedduetoaduodenalstricture.Thepatient remainsasymptomaticduringfollow-up.
Discussion: Groovepancreatitisisabenigncauseofobstructivejaundice,whosemain differ-entialdiagnosisisduodenalorpancreaticneoplasia.Whenthisconditioncausesduodenalor biliarystricture,surgicaltreatmentcanbenecessary.
© 2015 Sociedade Portuguesa de Gastrenterologia. Published by Elsevier España, S.L.U. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
∗Correspondingauthor.
E-mailaddress:ms18498@gmail.com(M.Gravito-Soares).
http://dx.doi.org/10.1016/j.jpge.2015.09.001
2341-4545/©2015SociedadePortuguesadeGastrenterologia.PublishedbyElsevierEspaña,S.L.U.Thisisanopenaccessarticleunderthe CCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).
IcteríciaObstrutiva; Obstruc¸ãoDuodenal; Pancreatite
Resumo
Introduc¸ão:A pancreatitedagoteiraduodeno-pancreática éumaformararadepancreatite crónica,queafetaaáreaanatómicaentreacabec¸adopâncreas,duodenoeductobiliarcomum. CasoClínico:Doente dosexo masculino, 67 anos, com antecedentes de cólicasbiliares de repetic¸ãoeconsumoetílicode30-40g/dia,internadoporicteríciaeprurido.Analiticamente, apresentavacolestasee,ecograficamente,dilatac¸ãomoderadadasviasbiliaresintra-hepáticas (VBIH),dilatac¸ãoquísticado1/3médiodocolédoco,ectasiadoWirsungeatrofiapancreática. Acolangiopancreatografiaporressonânciademonstrouaspetosimagiológicoscompatíveiscom pancreatiteparaduodenal.Aendoscopiaaltaexcluiuneoplasiaduodenal.Foisubmetidoa colan-giojejunostomiaemYRouxporestenosedocolédocoeapós5mesesagastrojejunostomiapor estenoseduodenal.Odoentemantemseguimento,permanecendoassintomático.
Discussão: Apancreatiteparaduodenaléumaformabenignadeicteríciaobstrutiva,cujo prin-cipaldiagnósticodiferencialéaneoplasiaduodenal/pancreática.Quandoestacondic¸ãocausa estenoseduodenaloubiliar,aterapêuticacirúrgicapoderásernecessária.
© 2015 Sociedade Portuguesa de Gastrenterologia. Publicado por Elsevier España, S.L.U. Este é um artigo Open Access sob a licença de CC BY-NC-ND (http://creativecommons.org/licenses/by-nc-nd/4.0/).
1.
Introduction
Groove pancreatitis is a rare and under-recognized sub-type of chronic pancreatitis that affects the anatomical region composed by the duodenum, pancreatic cephalic portionandcommonbiliaryduct(CBD).1---3 Theprevalence
ofthisconditioninpancreaticoduodenectomyspecimensof patientswithchronicpancreatitishasbeenreportedtobe ashighas24.5%.4Pathogenesisisstillunclear,butitinvolves
afibroticinflammation,5thatcouldcausebiliarystricture,
withobstructivejaundiceor duodenalstricturealongwith vomiting.The maindifferentialdiagnosisisthepancreatic head, CBD or duodenal neoplasia, which imply different managementandprognosis. Commonly,thiscondition can beresolvedwithconservative treatment. Inrare casesof clinicalrelevantbiliaryorduodenalstenosis,surgical treat-mentisnecessary.2,6
2.
Clinical
case
Theauthorspresentacaseofa67-year-oldman,witha his-toryofrepeatedbiliarycolic.Hehadanalcoholconsumption of30---40g/dayandrefusedcholecystectomy.Hepresented tothe emergency department with a two days evolution of painless jaundice, dark colouration of the urine and generalizedpruritus.Hedeniedhavinghadanyblood trans-fusion, newdrugs prescription or over-the-counter drugs, favabeanconsumption, previous surgeries, recenttravels orunprotectedsexualintercourse.Hehadnoprevious jaun-diceepisodesorrecentinfections.Thephysicalexamination revealedjaundiceofmucosaandskin.Theremaining phys-icalexamwasunremarkable.
Laboratory analysis showed predominant cholestatic hepatitis with aspartate aminotransferase 908 (<35U/L), alanine aminotransferase 1236 (<45U/L), alkaline
phosphatase 1581 (40---150UL), gamaglutamyl-transpeptidase 2753 (<55U/L), total bilirubin 17.2 (0.3---1.2mg/dL) and direct bilirubin 9.9 (0.1---0.5mg/dL) withnormalamylaseandlipase.Subsequentcomplementary studiesshowednoabnormalities,includingtumourmarkers carcinoembryonic antigen andcarbohydrate antigen 19-9, viral infections (coxsackie, hepatitis B, cytomegalovirus, varicella-zoster virus, herpes simplex virus, epstein---barr virus,humanimmunodeficiency virus),bacterialinfections (mycoplasma,legionella,leptospira,coxiellaburnetii, rick-ettsiaconorii,treponemapallidum, borreliaburgdorferi),
autoimmunityandserumIgG4.
The abdominal ultrasound showed a dilatation of the intra-hepatic biliary ducts (IHBD), middle third of CBD (16mm)andWirsung(7mm),withnormaldistalCBD diam-eter. Biliary lithiasis and pancreatic atrophy were also detected, but it wasnotpossible toidentify the obstruc-tive cause. The abdominal computerized tomography and themagnetic-resonancecholangiopancreatographyshowed IHBD dilatation, dilatation of proximal third of CBD mea-suring 17mm with progressive distal narrowing, pancreas divisum, ectasia of the duct of Wirsung with maximum diameter of 8mm, atrophy of pancreatic body and tail andcysticstructuresinpancreaticoduodenalgroove.These imagiological findingsappeared tohave been caused by a thick duodenal parietal wall at minor papilla level with consequent Wirsung obstruction and significant extrinsic compression of CBD which associated to the small cystic images in paraduodenal groove are strongly suggestive of groovepancreatitis(Fig.1).
Subsequently,anesophagogastroduodenoscopywas per-formed,revealinganasymptomaticduodenalstricturewith congestiveandinfiltrativemucosa,makingatranspapillary approachbyendoscopicretrograde cholangiopancreatogra-phy impossible. The biopsiesshowed moderateduodenitis withoutdysplasiaorneoplasia.
Figure1 Magneticresonancecolangio-pancreatographyimagesshowingcysticstructuresinduodenopancreaticgroove(a,b,d ande),pancreasdivisum(a---c)andenlargedWirsung(f).
Figure2 Computerizedtomographyscanshowingamarkedlyesophagealandgastricdilatation.
Despite conservative treatment with bowel rest, fluid therapy, analgesics, proton pump inhibitors and pancre-aticenzymesupplement,apersistentimportantcholestasis (total bilirubin ∼16mg/dL) and consequent obstructive jaundiceoccurred.Aftermultidisciplinaryconsultationthe patientwassubmittedtoaY-en-Rouxcholangiojejunostomy and cholecystectomy. Surgical specimen showed chronic lithiasiccholecystitis,cholestatichepatitisandCBDwithno abnormalities.
Fivemonthsafterthesurgicalintervention,thepatient returned to the emergency department due to recurrent vomiting and food intolerance. The ultrasound scan was normal in regards to IHDB and CBD. The plain abdomi-nal X-ray and computerized tomography revealed marked oesophageal and gastric dilatation with stasis (Fig. 2). An esophagogastroduodenoscopy confirmed the presence of severe esophagitis in relation to the gastroesophageal refluxofgastricstasiscontentduetoimpassableduodenal stricture. Biopsies continued to show moderate duodeni-tiswithoutdysplasiaorneoplasia.Thepatientwastreated withproton-pumpinhibitors,antacidsandprokineticswith noclinicalimprovement.Itwasthendecidedtosubmitthe
patienttoagastrojejunostomy.Atthetimeofthissurgical procedureanincisionalbiopsyofanteriorandlowerborder of pancreaswas also performed. Histopathology revealed chronicpancreatitiswithoutdysplasiaorneoplasia(Fig.3). Presently, the patient is asymptomatic and is being fol-lowedupinanoutpatientsurgicalclinic.Thepatientgave informedconsentforalldiagnosticandtherapeutic proce-dures.
3.
Discussion
Groovepancreatitisisanuncommonsubtypeoffocalchronic pancreatitisandararecauseofobstructivejaundice.Only averysmallnumberofcaseshavebeenpublished.1---3
The prevalence isunderestimated, butin surgical case seriesgroovepancreatitiswasdiagnosedin2---24.5%of pan-creaticoduodenectomyspecimensof patientswithchronic pancreatitis.4,7,8 Thiscondition isoftenpresent in
middle-agedmales(40---50yearsold)withalcoholconsumption.1,7---9
Theclinicalpresentationissimilartomoreprevalentforms of chronic pancreatitis, but recurrent vomiting due to
Figure3 Incisional biopsy ofanterior andlower border of headofthepancreasshowingfibrousandadipocytetissue,mild inflammationandneovascularizationinvolvingatrophic pancre-aticducts,suggestingchronicpancreatitis.
duodenalstrictureandimpairedmotilitytendstobemore frequent in groove pancreatitis.2 However, the majority
of cases had a mild CBD dilatation, insufficient to cause obstructivejaundice.9
Laboratoryanalysisoftenrevealsanormalorslight ele-vation of serum pancreatic enzymes and occasionally of serumliverenzymes.1,10 Tumourmarkersof
carcinoembry-onic antigen and carbohydrate antigen 19.9 are usually negative.1,8
Thediagnosisisdifficulttoestablishandinvolvesclinical, radiologicalandhistopathologicalfeatures.1Thecrucial
dif-ferentialdiagnosisisaheadpancreasadenocarcinoma,since themanagementandprognosisaredifferent.1,2,9,11Magnetic
resonanceis moresensible andspecifictodetectfindings, themostcommonbeingasheet-likemassbetweenthe pan-creasheadandduodenumwithduodenalwallcysticchanges andthickening.6,10
Inthehistopathologyanalysis,themacroscopicaspects are either an absent or narrow Santorini duct, or the presence of pancreas divisum and groove lesions similar to those of chronic pancreatitis.12,13 The duodenal wall
containsenlargedducts,inthemajorityofcaseswith thick-ened secretions, pseudocystic changes, adjacent stromal reactionsand Brunner glandhyperplasia.Fibrosisintothe adjacentpancreasandthesurroundingfatoccurs,especially inthegrooveareawhichinvolvestheCBD.14
Thepathogenesisisstillunclear,butthemostaccepted mechanism is a disturbance of the pancreatic outflow in the Santoriniduct through the minor papilla. One of the mainfactorsisprobablychronicalcoholconsumptionwhich increasestheviscosityofthepancreaticjuiceandleadsto Brunnerglandhyperplasia,causingocclusionordysfunction oftheminorpapilla.1,9,15,16
The treatment for acute phase of groove pancreatitis is conservative, including bowel rest, analgesia and flu-idstherapy.10 Inthechronic stagesoftheprocess,fibrosis
dominates over inflammation, and at this point surgical managementcouldbenecessary becauseofpersistent bil-iary or duodenal stricture.17 The surgical treatment of
and pathology features was used to make a diagnosis of groove pancreatitis in a middle-aged male with his-tory ofalcohol consumption.Although rare,this condition should be part of differential diagnosis of obstructive jaundice and an extensive evaluation should be carried out to exclude pancreatic, duodenal or CBD neoplasia. This case was initially complicated by biliary stricture and later by duodenal stricture, with the option for a less-invasive surgical intervention after malignancy being excluded.
Conflicts
of
interest
Theauthorshavenoconflictsofinteresttodeclare.
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