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CASEREPORTOpenAccessArarepresentationofPulmonaryLymphangiticCarcinomatosisinCancerofLip:CaseReportSajithBabu1*,SatheeshanB1,GeethaM2andSurijSalih1AbstractSquamouscellcarcinomaoflipisacommonmalignancyinIndiansubcontinent.
Metastaticspreadisinfrequent.
Althoughadvancedtumoursspreadtolymphnodesintheneck,itdoesnottypicallypresentwithlungmetastasisorwithlymphangiticcarcinomatosis.
Wedescribeapatientwhodevelopedcoughandincreasingdyspnoeawhileontreatmentforcarcinomaoflip.
Chestx-rayandcomputedtomographywereconsistentwithlymphangiticcarcinomatosis.
Lymphangiticcarcinomatosisoccurswithmanydifferentprimarytumoursandcanrarelyoccurinoralcancers.
Thisisthefirstreportfromcarcinomaoflip.
BackgroundThecommonsiteofmetastasisfrommostofthesolidmalignanciesislung.
Theyusuallyappearasnodularlesionsinradiologicimages.
Insomepatients,metastasispresentswithinterstitialspreadanditisreferredtoasPulmonaryLymphangiticCarcinomatosis(PLC).
HeadandneckcancersveryrarelyhavelungmetastasisintheformofPLC.
Oropharyngealandhypopharyngealcan-cershavebeenreportedtohavesuchtypeofmetastasis[1].
CanceroflipisacommonmalignancyinIndiansubcontinentmainlyduetotobaccochewingandthatthesecancersaredetectedinearlystagesduetoitsvisi-blelocation,aspreadtolungisrareandtheyareoftypicalnodularmetastases.
PLChasnotbeenreportedtilldatefromlipcancersinEnglishliterature.
HerewereportacaseofPLCarisingfromcancerofthelowerlip.
CasePresentation60yearoldgentlemanwithnocomorbidillness,pre-sentedwithasquamouscellcarcinomaoflowerlip.
Afterevaluation,thiswasstagedasT4N2aM0,stageIVandwasmoderatelydifferentiatedsquamouscellcar-cinoma.
TheX-rayofthechestwaswithinnormallim-its.
Wideexcisionofthelesionandreconstructionwithadeltopectoralflapandaradicalneckdissectiononipsilateralsidewasdone.
Postoperativehistopathologywasmoderatelydifferentiatedsquamouscellcarcinoma(pT4N2a).
After4weeks,postoperativeadjuvantcon-currentchemoradiationwasstartedwithCisplatinandradiotherapyin2Gyperfraction.
Whileonradiother-apy,thepatientdevelopedseveredyspnoeaofacuteonset.
Therewasnohistoryofsimilarepisodeinthepastandhewasnotaknownpatientofchronicobstructivepulmonarydisease.
Hewasafebrileandtherewasnocoughorexpectoration.
Basichaematologi-calstudyrevealednormalhaemogram.
Clinicallyhewasdyspnoeic,tachypnoeicandwithtachycardia.
Onauscul-tationofthechest,therewasscatteredcracklesandoccasionalronchi.
Airentrywasequalonbothsides.
Hewasputonsymptomaticcareintheformofbronchodi-lators,antibioticsandnasaloxygen.
Possibilitiesconsid-eredwereacutebronchopneumoniaandPLC.
Chestradiographrevealedinterstitiallinearpatternfromthehilumtotheouterlungfields(Figure1)andKerley'sBlinesinbothlungssuggestingPLC.
AcomputerizedtomographywastakenwhichshowednodularseptalthickeninganditstronglysuggestedthediagnosisofPLC(Figure2).
PatientwasgivenfurthercoursesofchemotherapywithCisplatin,butwithnoimprovement.
Thepatientsuccumbedtodiseaseoneighteenthdayafterthestartofpulmonarysymptoms.
DiscussionLungmetastasisfrommalignanttumoursusuallypre-sentasnodularlesionsandrarelyasPulmonaryLym-phangiticCarcinomatosis(PLC).
PLCischaracterisedbydiffusespreadofmalignancyinthelung,causinginflam-mationofthelymphvessels.
Thefirstreportedcaseof*Correspondence:drsajith@gmail.
com1DepartmentofSurgicalOncology,MalabarCancerCentre,Thalassery,KeralaFulllistofauthorinformationisavailableattheendofthearticleBabuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77WORLDJOURNALOFSURGICALONCOLOGY2011Babuetal;licenseeBioMedCentralLtd.
ThisisanOpenAccessarticledistributedunderthetermsoftheCreativeCommonsAttributionLicense(http://creativecommons.
org/licenses/by/2.
0),whichpermitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycited.
PLCwasbyGabrielAndralin1829[2].
ThediffuselyinfiltratingpatternofmetastasisasseeninPLCoccursin6-8%oflungmetastases[3].
80%ofthemarefromadenocarcinomas.
ThecommonsitesofprimaryfromwhichPLCoccursarecancersofbreast,bronchus,andstomach[4,5].
TheotherdescribedsiteswithPLCarecancersfromcolon,pancreas,kidney,cervix,thyroid,larynxandhypopharynx[6-8].
Thecancersofheadandneckrarelyshowthistypeofmetastasis.
Theexactreasonisunknown.
Thedescribedsitesinheadandneckregionarelarynx,hypopharynxandthyroid.
Metastasistolymphnodesfromadvancedcancersoflipisseeninabout44%.
Metastasistolungisreportedtobeverylow.
ThereisnoavailablereportsuggestingaPLCfromoralcancers.
PLCasmetastaticfeatureasseeninthecasedescribedinthismanuscriptisanextremelyrarepresentation.
Thepathophysiologyisthatthetumoursspreadbyhaematogenousroutetothelungandthenthroughthelymphaticswithinthelung.
Thelymphaticsinthelungareseenintheperibronchovascular,centrilobular,inter-lobularandsubpleuralregions.
Thetumourobstructstheselymphaticchannels.
Thedilatedlymphaticvesselsduetooedemafluid,tumoursecretionandthedesmo-plasticreactionbythetumourcells,producesinterstitialthickeningwhichisseenasstreaksinimagingstudies.
Thenodularpatternisduetothespreadoftumourintothelungparenchymaasseeninusuallungmetastases.
TheclinicalfeaturesofPLCaredyspnoeaandnonpro-ductivecoughwithcrepitationsandwithoutfeaturesofconsolidation.
ChestX-rayshowsseptallines(KerleyAandBlines).
Thedifferentialdiagnosisisinterstitiallungdisease,primarymalignancyinthelung,pulmonarysar-coidosisandhypersensitivitypneumonitis.
HRCTisthemodalityofchoiceforconfirmationofthediagnosis.
ThefindingsinCTscanare-thickeningofinterlobularsepta,fissuresandbronchovascularbundles.
Thesefind-ingsmaybeseenaslimitedordiffuseandmayinvolveunilateralorbilaterallungs.
Theradiologicpicturemaybesymmetricorasymmetricinbothlungs.
Theotherfindingsarenodularityinpleuraandgroundglassopa-city[9].
Thepossibilityofinterstitiallungdiseaseistobeconsideredandruledout.
PrakashPetaldescribedtheuseofPET/CTindiagnosingPLC.
Inastudyof35,theyfoundthatPET/CThashighspecificityindetectionofpulmonarylymphangiticcarcinomatosis[10].
Histopathologicalexaminationsshowinterstitialoedemaandfibrosisalongwithmalignantcellsandarefoundusuallyonpostmortembiopsy.
Sincetheradiolo-gicalfindinginapatientwithmalignantdiseaseelse-whereissuggestive,abiopsyofthelungisnotmandatory.
PLCoftenpresentsinthelatestagesofmalignancyanditindicatespoorprognosis.
ThetreatmentoptioninPLCiswithchemotherapy.
Cisplatinhavebeenfoundtobeeffective[11].
ConclusionPulmonaryLymphangiticCarcinomatosismayalsooccurrarelyinpatientswithoralcancersasseeninourpatientanditsprognosisisverypoorevenwithtreat-mentwithchemotherapy.
Figure1CXR:ChestRadiographshowingseptallines.
Figure2CTScan:CTscanofthoraxshowingdiffuseandbilateralfindings.
Babuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77Page2of3ConsentWritteninformedconsentwasobtainedfromthepatientforpublicationofthiscasereportandaccompanyingimages.
AcopyofthewrittenconsentisavailableforreviewbytheEditor-in-Chiefofthisjournal.
Authordetails1DepartmentofSurgicalOncology,MalabarCancerCentre,Thalassery,Kerala.
2DepartmentofRadiationOncology,MalabarCancerCentre,Thalassery,Kerala.
Authors'contributionsSBpreparedthemanuscriptandtheliteraturesearch,GMreviewedandeditedthemanuscript,STcorrectedandrevisedthemanuscript,SS:reviewedthemanuscript.
Allauthorsreadandapprovedthefinalmanuscript.
CompetinginterestsTheauthorsdeclarethattheyhavenocompetinginterests.
Received:8March2011Accepted:14July2011Published:14July2011References1.
ZieskeLA,MyersEN,BrownBM:Pulmonarylymphangiticcarcinomatosisfromhypopharyngealadenosquamouscarcinoma.
HeadNeckSurg1988,10(3):195-8.
2.
DoyleL:GabrielAndral(1797-1876)andthefirstreportsoflymphangitiscarcinomatosa.
JRSocMed1989,82(8):491-3.
3.
BruceDM,HeysSD,EreminO:Lymphangitiscarcinomatosa:aliteraturereview.
JRCollSurgEdinb1996,41(1):7-13.
4.
YamagishiY,AkibaY,IzumiyaM,HiguchiH,IizukaH,TakaishiH,NagataH,HibiT:[AcaseofadvancedgastriccancerwithlymphangitiscarcinomatosaafteroperationofKrukenbergtumortreatedbyTS-1plusCPT-11asthird-linechemotherapy].
GanToKagakuRyoho2005,32(8):1167-70.
5.
GuptaPR,JoshiN,MeenaRC,AliM:Asymptomaticlymphangitiscarcinomatosisduetosquamouscelllungcarcinoma.
IndianJChestDisAlliedSci2005,47(2):121-3.
6.
ThomasA,LenoxR:Pulmonarylymphangiticcarcinomatosisasaprimarymanifestationofcoloncancerinayoungadult.
CMAJ2008,179(4):338-40.
7.
KirkJE,KumaranM:Lymphangitiscarcinomatosaasanunusualpresentationofrenalcellcarcinoma:acasereport.
JMedCaseReports2008,2:19.
8.
YangSP,LinCC:[Pulmonarylymphangiticcarcinomatosis].
TaiwanYiXueHuiZaZhi1968,67(9):361-74.
9.
ZhangK,HuangY:[Clinicalfeaturesanddiagnosisofpulmonarylymphangiticcarcinomatosis].
AiZheng2006,25(9):1127-30.
10.
PrakashP,KalraMK,SharmaA,ShepardJA,DigumarthySR:FDGPET/CTinAssessmentofPulmonaryLymphangiticCarcinomatosis.
AJRAmJRoentgenol2010,194(1):231-6.
11.
KikuchiN,ShiozawaT,IshiiY,SatohH,NoguchiM,OhtsukaM:ApatientwithpulmonarylymphangiticcarcinomatosissuccessfullytreatedwithTS-1andcisplatin.
InternMed2007,46(8):491-4.
doi:10.
1186/1477-7819-9-77Citethisarticleas:Babuetal.
:ArarepresentationofPulmonaryLymphangiticCarcinomatosisinCancerofLip:CaseReport.
WorldJournalofSurgicalOncology20119:77.
SubmityournextmanuscripttoBioMedCentralandtakefulladvantageof:ConvenientonlinesubmissionThoroughpeerreviewNospaceconstraintsorcolorgurechargesImmediatepublicationonacceptanceInclusioninPubMed,CAS,ScopusandGoogleScholarResearchwhichisfreelyavailableforredistributionSubmityourmanuscriptatwww.
biomedcentral.
com/submitBabuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77Page3of3
Metastaticspreadisinfrequent.
Althoughadvancedtumoursspreadtolymphnodesintheneck,itdoesnottypicallypresentwithlungmetastasisorwithlymphangiticcarcinomatosis.
Wedescribeapatientwhodevelopedcoughandincreasingdyspnoeawhileontreatmentforcarcinomaoflip.
Chestx-rayandcomputedtomographywereconsistentwithlymphangiticcarcinomatosis.
Lymphangiticcarcinomatosisoccurswithmanydifferentprimarytumoursandcanrarelyoccurinoralcancers.
Thisisthefirstreportfromcarcinomaoflip.
BackgroundThecommonsiteofmetastasisfrommostofthesolidmalignanciesislung.
Theyusuallyappearasnodularlesionsinradiologicimages.
Insomepatients,metastasispresentswithinterstitialspreadanditisreferredtoasPulmonaryLymphangiticCarcinomatosis(PLC).
HeadandneckcancersveryrarelyhavelungmetastasisintheformofPLC.
Oropharyngealandhypopharyngealcan-cershavebeenreportedtohavesuchtypeofmetastasis[1].
CanceroflipisacommonmalignancyinIndiansubcontinentmainlyduetotobaccochewingandthatthesecancersaredetectedinearlystagesduetoitsvisi-blelocation,aspreadtolungisrareandtheyareoftypicalnodularmetastases.
PLChasnotbeenreportedtilldatefromlipcancersinEnglishliterature.
HerewereportacaseofPLCarisingfromcancerofthelowerlip.
CasePresentation60yearoldgentlemanwithnocomorbidillness,pre-sentedwithasquamouscellcarcinomaoflowerlip.
Afterevaluation,thiswasstagedasT4N2aM0,stageIVandwasmoderatelydifferentiatedsquamouscellcar-cinoma.
TheX-rayofthechestwaswithinnormallim-its.
Wideexcisionofthelesionandreconstructionwithadeltopectoralflapandaradicalneckdissectiononipsilateralsidewasdone.
Postoperativehistopathologywasmoderatelydifferentiatedsquamouscellcarcinoma(pT4N2a).
After4weeks,postoperativeadjuvantcon-currentchemoradiationwasstartedwithCisplatinandradiotherapyin2Gyperfraction.
Whileonradiother-apy,thepatientdevelopedseveredyspnoeaofacuteonset.
Therewasnohistoryofsimilarepisodeinthepastandhewasnotaknownpatientofchronicobstructivepulmonarydisease.
Hewasafebrileandtherewasnocoughorexpectoration.
Basichaematologi-calstudyrevealednormalhaemogram.
Clinicallyhewasdyspnoeic,tachypnoeicandwithtachycardia.
Onauscul-tationofthechest,therewasscatteredcracklesandoccasionalronchi.
Airentrywasequalonbothsides.
Hewasputonsymptomaticcareintheformofbronchodi-lators,antibioticsandnasaloxygen.
Possibilitiesconsid-eredwereacutebronchopneumoniaandPLC.
Chestradiographrevealedinterstitiallinearpatternfromthehilumtotheouterlungfields(Figure1)andKerley'sBlinesinbothlungssuggestingPLC.
AcomputerizedtomographywastakenwhichshowednodularseptalthickeninganditstronglysuggestedthediagnosisofPLC(Figure2).
PatientwasgivenfurthercoursesofchemotherapywithCisplatin,butwithnoimprovement.
Thepatientsuccumbedtodiseaseoneighteenthdayafterthestartofpulmonarysymptoms.
DiscussionLungmetastasisfrommalignanttumoursusuallypre-sentasnodularlesionsandrarelyasPulmonaryLym-phangiticCarcinomatosis(PLC).
PLCischaracterisedbydiffusespreadofmalignancyinthelung,causinginflam-mationofthelymphvessels.
Thefirstreportedcaseof*Correspondence:drsajith@gmail.
com1DepartmentofSurgicalOncology,MalabarCancerCentre,Thalassery,KeralaFulllistofauthorinformationisavailableattheendofthearticleBabuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77WORLDJOURNALOFSURGICALONCOLOGY2011Babuetal;licenseeBioMedCentralLtd.
ThisisanOpenAccessarticledistributedunderthetermsoftheCreativeCommonsAttributionLicense(http://creativecommons.
org/licenses/by/2.
0),whichpermitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycited.
PLCwasbyGabrielAndralin1829[2].
ThediffuselyinfiltratingpatternofmetastasisasseeninPLCoccursin6-8%oflungmetastases[3].
80%ofthemarefromadenocarcinomas.
ThecommonsitesofprimaryfromwhichPLCoccursarecancersofbreast,bronchus,andstomach[4,5].
TheotherdescribedsiteswithPLCarecancersfromcolon,pancreas,kidney,cervix,thyroid,larynxandhypopharynx[6-8].
Thecancersofheadandneckrarelyshowthistypeofmetastasis.
Theexactreasonisunknown.
Thedescribedsitesinheadandneckregionarelarynx,hypopharynxandthyroid.
Metastasistolymphnodesfromadvancedcancersoflipisseeninabout44%.
Metastasistolungisreportedtobeverylow.
ThereisnoavailablereportsuggestingaPLCfromoralcancers.
PLCasmetastaticfeatureasseeninthecasedescribedinthismanuscriptisanextremelyrarepresentation.
Thepathophysiologyisthatthetumoursspreadbyhaematogenousroutetothelungandthenthroughthelymphaticswithinthelung.
Thelymphaticsinthelungareseenintheperibronchovascular,centrilobular,inter-lobularandsubpleuralregions.
Thetumourobstructstheselymphaticchannels.
Thedilatedlymphaticvesselsduetooedemafluid,tumoursecretionandthedesmo-plasticreactionbythetumourcells,producesinterstitialthickeningwhichisseenasstreaksinimagingstudies.
Thenodularpatternisduetothespreadoftumourintothelungparenchymaasseeninusuallungmetastases.
TheclinicalfeaturesofPLCaredyspnoeaandnonpro-ductivecoughwithcrepitationsandwithoutfeaturesofconsolidation.
ChestX-rayshowsseptallines(KerleyAandBlines).
Thedifferentialdiagnosisisinterstitiallungdisease,primarymalignancyinthelung,pulmonarysar-coidosisandhypersensitivitypneumonitis.
HRCTisthemodalityofchoiceforconfirmationofthediagnosis.
ThefindingsinCTscanare-thickeningofinterlobularsepta,fissuresandbronchovascularbundles.
Thesefind-ingsmaybeseenaslimitedordiffuseandmayinvolveunilateralorbilaterallungs.
Theradiologicpicturemaybesymmetricorasymmetricinbothlungs.
Theotherfindingsarenodularityinpleuraandgroundglassopa-city[9].
Thepossibilityofinterstitiallungdiseaseistobeconsideredandruledout.
PrakashPetaldescribedtheuseofPET/CTindiagnosingPLC.
Inastudyof35,theyfoundthatPET/CThashighspecificityindetectionofpulmonarylymphangiticcarcinomatosis[10].
Histopathologicalexaminationsshowinterstitialoedemaandfibrosisalongwithmalignantcellsandarefoundusuallyonpostmortembiopsy.
Sincetheradiolo-gicalfindinginapatientwithmalignantdiseaseelse-whereissuggestive,abiopsyofthelungisnotmandatory.
PLCoftenpresentsinthelatestagesofmalignancyanditindicatespoorprognosis.
ThetreatmentoptioninPLCiswithchemotherapy.
Cisplatinhavebeenfoundtobeeffective[11].
ConclusionPulmonaryLymphangiticCarcinomatosismayalsooccurrarelyinpatientswithoralcancersasseeninourpatientanditsprognosisisverypoorevenwithtreat-mentwithchemotherapy.
Figure1CXR:ChestRadiographshowingseptallines.
Figure2CTScan:CTscanofthoraxshowingdiffuseandbilateralfindings.
Babuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77Page2of3ConsentWritteninformedconsentwasobtainedfromthepatientforpublicationofthiscasereportandaccompanyingimages.
AcopyofthewrittenconsentisavailableforreviewbytheEditor-in-Chiefofthisjournal.
Authordetails1DepartmentofSurgicalOncology,MalabarCancerCentre,Thalassery,Kerala.
2DepartmentofRadiationOncology,MalabarCancerCentre,Thalassery,Kerala.
Authors'contributionsSBpreparedthemanuscriptandtheliteraturesearch,GMreviewedandeditedthemanuscript,STcorrectedandrevisedthemanuscript,SS:reviewedthemanuscript.
Allauthorsreadandapprovedthefinalmanuscript.
CompetinginterestsTheauthorsdeclarethattheyhavenocompetinginterests.
Received:8March2011Accepted:14July2011Published:14July2011References1.
ZieskeLA,MyersEN,BrownBM:Pulmonarylymphangiticcarcinomatosisfromhypopharyngealadenosquamouscarcinoma.
HeadNeckSurg1988,10(3):195-8.
2.
DoyleL:GabrielAndral(1797-1876)andthefirstreportsoflymphangitiscarcinomatosa.
JRSocMed1989,82(8):491-3.
3.
BruceDM,HeysSD,EreminO:Lymphangitiscarcinomatosa:aliteraturereview.
JRCollSurgEdinb1996,41(1):7-13.
4.
YamagishiY,AkibaY,IzumiyaM,HiguchiH,IizukaH,TakaishiH,NagataH,HibiT:[AcaseofadvancedgastriccancerwithlymphangitiscarcinomatosaafteroperationofKrukenbergtumortreatedbyTS-1plusCPT-11asthird-linechemotherapy].
GanToKagakuRyoho2005,32(8):1167-70.
5.
GuptaPR,JoshiN,MeenaRC,AliM:Asymptomaticlymphangitiscarcinomatosisduetosquamouscelllungcarcinoma.
IndianJChestDisAlliedSci2005,47(2):121-3.
6.
ThomasA,LenoxR:Pulmonarylymphangiticcarcinomatosisasaprimarymanifestationofcoloncancerinayoungadult.
CMAJ2008,179(4):338-40.
7.
KirkJE,KumaranM:Lymphangitiscarcinomatosaasanunusualpresentationofrenalcellcarcinoma:acasereport.
JMedCaseReports2008,2:19.
8.
YangSP,LinCC:[Pulmonarylymphangiticcarcinomatosis].
TaiwanYiXueHuiZaZhi1968,67(9):361-74.
9.
ZhangK,HuangY:[Clinicalfeaturesanddiagnosisofpulmonarylymphangiticcarcinomatosis].
AiZheng2006,25(9):1127-30.
10.
PrakashP,KalraMK,SharmaA,ShepardJA,DigumarthySR:FDGPET/CTinAssessmentofPulmonaryLymphangiticCarcinomatosis.
AJRAmJRoentgenol2010,194(1):231-6.
11.
KikuchiN,ShiozawaT,IshiiY,SatohH,NoguchiM,OhtsukaM:ApatientwithpulmonarylymphangiticcarcinomatosissuccessfullytreatedwithTS-1andcisplatin.
InternMed2007,46(8):491-4.
doi:10.
1186/1477-7819-9-77Citethisarticleas:Babuetal.
:ArarepresentationofPulmonaryLymphangiticCarcinomatosisinCancerofLip:CaseReport.
WorldJournalofSurgicalOncology20119:77.
SubmityournextmanuscripttoBioMedCentralandtakefulladvantageof:ConvenientonlinesubmissionThoroughpeerreviewNospaceconstraintsorcolorgurechargesImmediatepublicationonacceptanceInclusioninPubMed,CAS,ScopusandGoogleScholarResearchwhichisfreelyavailableforredistributionSubmityourmanuscriptatwww.
biomedcentral.
com/submitBabuetal.
WorldJournalofSurgicalOncology2011,9:77http://www.
wjso.
com/content/9/1/77Page3of3
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