Bilateral pyosalpinx in a peripubescent female with Hirschsprung’s disease: a case report

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Title:
Bilateral pyosalpinx in a peripubescent female with Hirschsprung’s disease: a case report
Series Title:
International Journal of Emergency Medicine
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Mixed Material
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English
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Desai, Bobby
Ward, Timothy
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BioMed Central
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Abstract:
This is a case report of bilateral pyosalpinx in a peripubescent female with a history of Hirschsprung’s disease. Bilateral pyosalpinx is a rare condition in non-sexually active females. The presence of this disease in a patient with a history of Hirschsprung’s disease is concerning for an association of the two processes.
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International Journal of Emergency Medicine 2011 4:64
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Publication of this article was funded in part by the University of Florida Open-Access publishing Fund. In addition, requestors receiving funding through the UFOAP project are expected to submit a post-review, final draft of the article to UF's institutional repository, IR@UF, (www.uflib.ufl.edu/ufir) at the time of funding. The Institutional Repository at the University of Florida (IR@UF) is the digital archive for the intellectual output of the University of Florida community, with research, news, outreach and educational materials

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University of Florida
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University of Florida
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All rights reserved by the source institution.
Resource Identifier:
doi - 10.1186/1865-1380-4-64
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AA00007505:00001


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CASEREPORT OpenAccessBilateralpyosalpinxinaperipubescentfemale withHirschsprung sdisease:acasereportBobbyDesai*andTimothyWardAbstractThisisacasereportofbilateralpyosalpinxinaperipubescentfemalewithahistoryofHirschsprung sdisease. Bilateralpyosalpinxisarareconditioninnon-sexuallyactivefemales.Thepresenceofthisdiseaseinapatientwith ahistoryofHirschsprung sdiseaseisconcerningforanassociationofthetwoprocesses.IntroductionThisisacasereportofbilateralpyosalpinxinaperipubescentfemalewithahistoryofHirschsprung sdisease. Bilateralpyosalpinxisarareconditioninnon-sexually activefemales.Thepresenceofthisdiseaseinapatient withahistoryofHirschsprung sdiseaseisconcerning foranassociationofthetwoprocesses.CaseReportA12yearoldfemalepresentstotheemergencydepartmentwithacomplaintofabdominalpain.Shehasa pastmedicalhistoryofHirschsprung sdiseasewitha stagedrepair.Atdayfouroflifesheunderwentcolostomywithresectionoftheaffectedcolonfromthemid transversecolontothejunctionofthesigmoidanddescendingcolon.Shereturnedtwomonthslaterforthe secondstageoftherepairwheresheunderwentaSoave endorectalpullthruprocedurewithincidentalappendectomy.Sincethattimeshehashadagoodrecovery withoutconstipationordiarrheaandwithnormalbowel function.Shehadrecurrenttonsillitisandunderwent tonsillectomy.Shetakesnomedications.Thepatient complainsofasharpbilaterallowerabdominalpainfor thepasttwodaysthatisgreatestinthesuprapubic region.Shehashadthreeepisodesofemesis.Shedenies achangeinbowelhabits.Shedoesreportalowgrade feverto101.Shedeniesdysuria,frequencyorhematuria. Shedeniesanyhistoryofsexualactivity.Herfirstmenstrualperiodwassixweeksagoandhersecondmenstrualperiodwastwoweeksago.Shecomplainsofa newwateryvaginaldischargeforlessthanoneday. Uponarrivalhervitalsaretemperature37.2degrees Celsiusbymouth,pulse125beatsperminute,blood pressure124/64mm/Hg,andpulseox96%onroomair. Sheweighs45kg.Sheisinobviousmoderatedistress duetoherpain.Herbowelsoundsarenormal.Her abdomenisnondistendedandfirmwithvoluntary guarding.Itisdiffuselytender,butworseinthebilateral lowerquadrantswithoutreboundtenderness.Thereis noCVAtenderness.Onpelvicexam,therearenormal externalgenitaliaTannerstageII-IIIwithintacthymen fromsixo clocktonineo clockposition.Thereareno obviousperinealorvaginallacerations.Awateryblood tingeddischargeispresent.Therestofherphysical examisunremarkable. Initiallabsshowedanormalmetabolicpanel.The completebloodcounthadanormalhemoglobinand hematocritwithawhitebloodcellcountof14.5thou/ cumm.Therewere64percentneutrophilsand18percentlymphocyteswith11percentbands.Herurinalysis had219redbloodcellsand85whitebloodcellswitha largeamountofsquamousepithelialcells.Itwasnitrite negativeandhadlargeleukocyteesterase.UrinePCR forgonorrheaandchlamydiawasnegative.Theurine pregnancytestwasnegative.CTscanoftheabdomen andpelviswithIVandoralcontrastshowednormal lungbases,liver,spleen,pancreas,gallbladder,kidneys, andadrenalglands.Therewerenobowelobstruction noted.Bilateraldilatedtubularstructureswerenotedin thelowerquadrants,adnexalregions,withwallenhancementandsurroundinginflammatorychangesconsistent withbilateralpyosalpinx.Therewerenodistinctdrainableabscessesseen. SeeFigures1,2,3,4and5:CTscanoftheabdomen andpelviswithintravenousandoralcontrastshowing *Correspondence:bdesai@ufl.edu UniversityofFlorida,DepartmentofEmergencyMedicine,POBox100186, Gainesville,FL32610,USADesaiandWard InternationalJournalofEmergencyMedicine 2011, 4 :64 http://www.intjem.com/content/4/1/64 2011DesaiandWard;licenseeSpringer.ThisisanOpenAccessarticledistributedunderthetermsoftheCreativeCommons AttributionLicense(http://creativecommons.org/licenses/by/2.0),whichpermitsunrestricteduse,distribution,andreproductionin anymedium,providedtheoriginalworkisproperlycited.

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bilateraldilatedfallopiantubeswithpronouncedwall enhancement ShereceivedIVfluidsandmorphineforpaincontrol, andshewasadmittedtogynecologyserviceforIVantibiotics.InthehospitalshereceivedIVAmpicillin,Gentamicin,andFlagylforfourdaysuntilshewasafebrile forfortyeighthoursandhadanormalwhitecount.She wasdischargedonatendaycourseofDoxycyclineand FlagylwithMotrinforpaincontrol.Atsixmonthtelephonefollowupshedeniesanyrecurrenceofher symptoms.DiscussionThepresentationofa12yearold,whoisnotsexually active,withbilateralpyosalpinxandahistoryofHirschsprung sdiseaseisextremelyrare.Thepathologyand anatomicallocationofthesetwodiseasesprocessessuggestthattheymaybeassociated. Hirschsprungdiseaseisanuncommoncongenitaldisorderthataffects1in5000oflivebirths.Thediseaseis characterizedbytheabsenceofganglioncellsinthedistalcolonincludingtheanus.Itresultsfromincomplete migrationofneuralcrestcellsorearlycelldeath.There havebeeneightgenesidentifiedthatareassociatedwith Hirschsprungdiseaseandthediseasehasbeenassociatedwithothercongenitalabnormalities.Fivepercent ofallcasesareassociatedwithDownsyndrome[1].In oneprospectivestudy25percentofHirschsprung patientswerealsofoundtohavecongenitalabnormalitiesofthekidneyandgenitaltrack,themostcommon abnormalitiesbeinghydronephrosisandhypoplasia[2]. TheassociationofhydrosalpinxandHirschsprung s diseasewaspreviouslysuggestedin2007byMerliniet al.Inthiscaseseriestheauthorsuggestthatbilateral hydrosalpingesisaassociatedabnormalityofHirschsprung sdiseaseduetoneurocristopathy.Thepaperalso Figure1 CTscanoftheabdomenandpelviswithintravenous andoralcontrast .Bilateraldilatedfallopiantubeswithpronounced wallenhancementareshown. Figure2 CTscanoftheabdomenandpelviswithintravenous andoralcontrast .Swellingandinflammationareshownaround thefallopiantube. Figure3 CTscanoftheabdomenandpelviswithintravenous andoralcontrast .Inflammationisdemonstrated. Figure4 CTscanoftheabdomenandpelviswithintravenous andoralcontrast .AlowercutontheCTscandemonstrating extensionofinflammation. DesaiandWard InternationalJournalofEmergencyMedicine 2011, 4 :64 http://www.intjem.com/content/4/1/64 Page2of3

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discussesthepossibilitythatthediseaseprocesscould beacomplicationfromthesurgicalrepair.Thisisthe onlyarticlethatwasfoundinapubmedsearchfrom 1980-presentwhichdiscusseshydrosalpinxorpyosalpinxinassociationwithHirschsprungdisease[3]. Pyosalpinxistheacuteinflammationofthesalpinx whichismostcommonlycausedbygonorrhea.Other welldescribedcausesofpyo salpinxareChlamydiaand entericbacteria[1].Hydrosapinxhasbeenassociated withlesscommonorganismsincludingpneumococcus, streptococcus,andshigelloidesandisseeninnonsexuallyactivefemales[4-7].Bothpyosalpinxandhydrosalpinxhavebeenreportedtopresentatmenarchein femaleswithunderlyingurogenitalmalformations[8]. Inthiscasethepatientpresentedjustpriortohersecondmenstrualperiodwith pyosalpinxthatwasconfirmedbyCTexam.Shehadcompleteresolutionof symptomswithIVandPOantibioticsanddidnothave returnofsymptomsatsixmonthfollowup.ItisreasonabletospeculatethatherunderlyingHirschsprung sdiseaseattributedtothiscondition.ConsentWritteninformedconsentwasobtainedfromtheparentsofthepatientforpublicationofthisCasereport andanyaccompanyingimages.Acopyofthewritten consentisavailableforreviewbytheEditor-in-Chiefof thisjournal.Authors contributions TW&BKD:Wroteandeditedcasereport Competinginterests Theauthorsdeclarethattheyhavenocompetinginterests. Received:5May2011Accepted:12October2011 Published:12October2011 References1.KumarV,AbbasAK,FaustoN: RobbinsandCotranPathologicBasisof Disease. ElsevierSaunders;,7830-831,1064-1065. 2.PiniPratoA,MussoM,CeccheriniI,MattioliG,GiuntaC,GhiggeriGM, JasonniV: Hirschsprungdiseaseandcongenitalanomaliesofthekidney andurinarytract(CAKUT):anovelsyndromicassociation. Medicine (Baltimore) 2009, 88(2) :83-90. 3.MerliniL,AnooshiravaniM,PeiryB,LaScalaG,HanquinetS: Bilateral hydrosalpinxinadolescentgirlswithHirschsprung sdiseaseassociationoftworareconditions. AJR 2008, 190 :278-282. 4.CasiroOG,GochbergF,DrachmanR: Pneumococcalpyosalpinxina prepubertalchild. IsrJMedSci 1980, 16(12) :865-6. 5.HornemannA,vonKoschitzkyH,BohlmannMK,HornungD,DiedrichK, TaffazoliK: Isolatedpyosalpinxina13-year-oldvirginFertilSteril. 2009, 91(6) :2732.e9-10. 6.RothT,HentschC,ErardP,TschantzP: Pyosalpinx:notalwaysasexual transmitteddisease?PyosalpinxcausedbyPlesiomonasshigelloidesin animmunocompetenthost. ClinMicrobiolInfect 2002, 8(12) :803-5. 7.MeisJF,FestenC,Hoogkamp-KorstanjeJA: Pyosalpinxcausedby Streptococcuspneumoniaeinayounggirl. PediatrInfectDisJ 1993, 12(6) :539-40. 8.MollittDL,SchullingerJN,SantulliTV,HensleTW: Complicationsat menarcheofurogenitalsinuswithassociatedanorectalmalformations. J PediatrSurg 1981, 16(3) :349-52.doi:10.1186/1865-1380-4-64 Citethisarticleas: DesaiandWard: Bilateralpyosalpinxina peripubescentfemalewithHirschsprung sdisease:acasereport. InternationalJournalofEmergencyMedicine 2011 4 :64. Submit your manuscript to a journal and bene t from:7 Convenient online submission 7 Rigorous peer review 7 Immediate publication on acceptance 7 Open access: articles freely available online 7 High visibility within the eld 7 Retaining the copyright to your article Submit your next manuscript at 7 springeropen.com Fi gu r e 5 CT s ca n of t he ab do m en an d pe lv is w it h i nt r av e no us and oral contrast Abscess is shown. Desai and Ward International Journal of Emergency Medicine 2011, 4 :64 http://ww w.intjem.com/c ontent/4/1/64 Page 3 of 3