Vulvoperineal Crohn's disease responsive to metronidazole 2013 Aristóteles Rosmaninho.pdf

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CASEREPORT
s
VulvoperinealCrohn´sdiseaseresponsivetometronidazol*
DoençadeCrohnvulvoperinealresponsivaaometronidazole
AristótelesRosmaninho1MadalenaSanches2MartaSalgado3
RosárioAlves1ManuelaSelores4
DOI:/abd1806-
Abstract:Crohn´sdiseaseisamultisystemchronicgranulomatousinflammatorydiseasethatprimarilyaffects
,thecutaneousmanifestationsfollowtheintestinaldisease,
butoccasionallydermatologicallesionsaretheinauguraleventandmayconstitutetheonlysignofthedisease.
Vulvoperinealinvolvementisrare,mayprecedebowelsymptomsbymonthstoyearsandmaygounrecognized.
DuetothepaucityofreportsofCrohn´sdiseaseatthislocationandintheabsenceofrandomizedtrials,thereare
-oldwomanwithvulvope-
rinealCrohn´sdiseasewithoutdigestiveinvolvement,thatwassuccessfullymanagedwithmetronidazole.
Keywords:Crohndisease;Metronidazole;Therapeutics
Resumo:AdoençadeCrohnéumadoençagranulomatosamultissistêmicainflamatóriacrónicaqueafectapri-
,asmanifestaçõescutâneassucedemadoençaintesti-
nal,mas,ocasionalmente,aslesõesdermatológicassãooprimeiroeventoepodemconstituiroúnicosinalda
doençéraro,podeprecederossintomasintestinaisemmesesouanos,epode
àescassezderelatosdedoençadeCrohncomestalocalizaçãoenaausênciade
ensaiosclínicosrandomizados,nãohánenhumtratamentopadrãoparaadoençacutâ
deumamulherde47anoscomdoençadeCrohnvulvoperinealsemenvolvimentodigestivo,quefoitratadacom
sucessocommetronidazol.
Palavras-chave:DoençadeCrohn;Metronidazol;Terapêutica
INTRODUCTION
Crohn´sdiseaseisamultisystemchronicgran-
ulomatousinflammatorydiseasethatprimarily(includingsurgery,useofantibioticsandimmunosup-
)
cases,cutaneousmanifestationsfollowtheintestinalareonlyanecdotalreportsthatsupporttheuseof
disease,-old
theinauguraleventandmayconstitutetheonlysignwomanwithvulvoperinealCrohn´sdiseasewithout
,maydigestiveinvolvementthatwassuccessfullymanaged


Crohn´sdiseaseatthislocationandintheabsenceofofvulvoperinealCrohn´sdiseaseandcutaneousflares
randomizedtrials,therearenostandardtreatmentsalsorespondrapidlytorepeatedtreatment.
.
.
*WorkperformedattheDermatologyService;HospitaldeSantoAntónio-HospitalCenteratPorto,–Porto,Portugal.
FinancialSupport:none
ConflictofInterests:none
1MD-HospitalAssistantofDermatovenereologyattheHospitaldeSantoAntónio–HospitalCenteratPorto,–Porto,Portugal.
2MD-GraduateAssistantofDermatovenereologyattheHospitaldeSantoAntónio–HospitalCenteratPorto,–Porto,Portugal.
3MD-HospitalAssistantofGastroenterologyattheHospitaldeSantoAntónio–HospitalCenteratPorto,–Porto,Portugal.
4MD-DirectoroftheDermatovenereologyServiceattheHospitaldeSantoAntónio-HospitalCenteratPorto,–Porto,Portugal.
©2013byAnaisBrasileirosdeDermatologia
;88(6Suppl1):S71-4.
72RosmaninhoA,SanchesM,SalgadoM,AlvesR,SeloresM
CASEREPORT(<5).Syphilis, HIV serologiesandMantouxtestwere
A47-year-oldwomanpresentedforevaluationnegative. PCRtestingforherpessimplexviruswas
-rayshowednoabnormalities.
-Basedontheclinicalandhistologicalfindingsavul-
´sdisease(CD)diagnosis
-
medicalhistorywasonlyremarkableforhyperten-
,anexuberantinflam-studiesofthedigestivetractandbiopsiesfromthe
mationofthevulvawithaftoidulcersofthelabiaileum,colonandrectumshowednoabnormalities,so
minorandmajora,andlinearulcerationsinthethatadiagnosisofvulvoperinealcutaneousCDwith-
-
sureabovetheclitorisanda“knife-cut”ulcerwithwentondailymetronidazole(1000mg)and
sharpmarginsonthebuttocksfoldwerealsopresentciprofloxacin(1000mg)withastrikingimprovement.
(Figure1).Askinbiopsyrevealedanulceratedepider-Antibiotherapywasdiscontinuedat3monthsand
miswithafistuloustractandaspectsofchronicazathioprine(50mgdaily),
inflammationwithlymphocytes,hystiocytesandtheconditionworsenedandmetronidazole(1000mg
multinucleatedgiantcellsina non-caseating granulo-daily)wasaddedtoazathioprineandmarked
matouspatternonthedermis(Figure2).
mycobacterialelementswerenotdetectedbyhistol-ofcombinedtherapy(metronidazole1000mgplus
)sustainedclinical
werenormalexceptforC-reactiveproteinof41mg/dlimprovementwasobserved(Figure3).
FIGURE1:Clinicalfin-
dingsatpresentation
FIGURE2:H&E(10x):
ulceratedepidermis
withafistuloustract
(left).PAS(40x):
aspectsofchronic
inflammationina
granulomatouspat-
terninthedermis
(right)
;88(6Suppl1):S71-4.
VulvoperinealCrohn´sdiseaseresponsivetometronidazole73
FIGURE3:
Clinicalimpro-
vementwith
metronidazole
andazathioprine
at2monthsof
therapy
DISCUSSION
InCDthemucocutaneouslesionsusuallyfol-resultsandinclude:topical,intralesionalandsystemic
lowtheintestinaldiseaseandexceptionallyoccurcorticosteroids,sulfasalazine,azathioprine,
,ahighcyclosporine,methotrexate,thalidomide,metronida-
indexofsuspicionmayberequiredtoreachtheaccu-zole,infliximab,adalimumabandsurgicalexcision.

extensionoftheintestinaldisease(,3,4-7Assupported
andfistula)andmorerarelytheyoccurinanoncon-byourcase,itseemstobeeffectivewithresolutionof
tiguousextraintestinalspread(metastaticCD).-
,3Andreani etal,%hasbeen
reportedthat25%ofvulvarCDdidnothaveanypre--
viousintestinalsymptomsandpatientshadnotbeeninflammatory,antibacterialandimmunosuppressive
, etal,thelesionsofall
andpainthatevolvetovulvarhypertrophy,ulceration26patientswithperinealCDcleared,andanoptimal
andabscessesarethetypicalclinicalpicture.“Knife-doseof20mg/kg/dayfor12to36monthswas
cut”ulcers(asinourcase)
ofCD,althoughtheyhavebeendescribedincuta-recurrences,howevercutaneouslesionspromptly
neoustuberculosisandherpeticinfectionsinrespondtorepeatedtreatmentwiththe
,8Reversibleparesthesiaisthemajorcomplica-
examinationcharacteristicallyshowsnon-caseatingtionreportedwithlong-termmetronidazolethera-

granulomatousdiseases thatcancausevulvarulcera-darkeningoftheurinetogetherwithgastrointestinal
tion suchassarcoidosis,tuberculosis,fungalinfec--tolerated
tions, lymphogranulomavenereum,granulomawithoutanysideeffects. Wereportanextremelyatyp-

gangrenosumcanclinicallymimiccutaneousCrohn´swithmetronidazolewithnosignificantadverse
--
ologicalandmycologicalworkup. Duetoapaucityoftionasitmayprecedegastrointestinalinvolvement
reportswiththistopographythetreatmentremainsandbeacauseofhighmorbidityifnotpromptlyrec-

cutaneousdiseasehavebeenproposedwithvariable
;88(6Suppl1):S71-4.
74RosmaninhoA,SanchesM,SalgadoM,AlvesR,SeloresM
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Howtocitethisarticle:RosmaninhoA,SanchesM,SalgadoM,AlvesR,´sdisea-
;88(6Suppl1):S71-4.
;88(6Suppl1):S71-4.