CongenitaldeficiencyofmeibomianglandsOBLIQUEGLANDSFig1DiagramtoillustratethedispositionofresidualmeibomianglandsINCOMPLETEGLANDRUDIMENTSfromanAmericanAirBasewithahistoryofoneandahalfmonthsofsoftcont ID: 944066
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BritishJournalofOphthalmology,1987,71,312-314CongenitaldeficiencyofmeibomianglandsAJBRONANDLSMENGHERFromtheNuffieldLaboratoryofOphthalmology,UniversityofOxford,WaltonStreet,OxfordOX26AWSUMMARYA16-year-oldgirlpresentedwithcontactlensintolerance.Shewasfoundtohaveamarkeddeficiencyofmeibomianglandsintheupperlidsandalmosttotalabsenceinthelowerlids.Evidenceoftearfilminstabilitywasfoundandattributedtodeficientlidoilproduction.Adailywearsoftcontactlenswaslaterfittedandtolerated.Meibomianlipidformstheanteriorlayerofthetearfilm.'Itisbelievedtoretardevaporationfromthepreoculartearfilmandtopreventcontaminationofthetearfilmbythemorepolarskinsebumofthecutaneoussurfaceofthelidmargin.Meibomianlipidissecretedbythemeibomianglands,whichareradiallydisposedandseriallyarrangedwithinthesubstanceofthetarsalplate,withtheirductularopeningsplacedonthecutaneoussideofthemucocutaneousjunction.Thisidealplacementpermitsthedeliveryofmeibomianoilontotheanterioraspectofthetearfilm,whereitspreadsovertheaqueousfilmastheeyesopenaftereachblink.Histologicallytheglandsareracemoseholocrineglands,whoseaciniopenintoacentralductulelyingatrightanglestothelidmargin.Theaciniformaseriesofyellowishgrape-likeclusters,distributedatintervalsalongthelengthofthecentralductuleandclearlyvisiblethroughtheconjunctivaofthetarsalplateinyoungadults.Withincreasingagetheseacinardetailsarelessvisible,thoughayellowstreakcanbeseenrepresentingeachgland.Theglandsarealsovisiblebytransilluminationthroughthelid,"andaninterestinginfraredtechnique(meibomo-scopy)hasbeendeveloped.Theductularorificesatthelidmarginarevisiblysurroundedbyacuffofepithelialtissue,whichhasadelicatevasculaturevisiblebyfluoresceinangio-graphy.3Theorificesareflushwiththesurfaceinhealthandarranged,usuallyinasingleline,parallelbutanteriortothemucocutaneousjunction.Attimesthelineislessregularandmaygivetheimpressionofaduplicationintheantero-posteriordirection.Theepithelialcuffisofsimilarpinkcolourtoneighbour-inglidmargintissue.Inblackpeopletheorificesareoutlinedbypigment,andaremorereadilyseen.CorrcspondencctoAJBron,FRCS.Therearemoreglandsintheupperthanthelowerlid(30-40intheupperand20-30inthelower).Gentlepressurethroughthelidontotheglobeimpressesthetarsalglandsandexpressesaclearfluidlipidontothelidmargin.Meibomianoilisliquidatlidtemperature(withanuppermeltingpointrangeat32*0-338'C).6Thestimulusforsecretionofmeibomianlipidisnotknown.Althoughdiseaseofthemeibomianglandsiscommonaspartofmeibomitis,inassociationwithmarginalblepharitis,orwithchalazia,orrosacea,therearefewreportsrelatingsuchabnormalitytosurfaceoculardisease.However,variousauthors'-"'referredtoconjunctivitisaccompanyingmeibomianglanddisease,andBronandTripathi(1973)toaspecificformofepithelialkeratopathycausedbymeibomianitis(cysticepithelialkeratopathy)."Thygeson'2consideredtheroleofstaphylococcaltoxinsinblepharoconjunctivitis.EpithelialkeratitishasbeendescribedbyMcCulleyandSciallisassoci-atedwithmeibomianitis.'3Ithasbeenassumedthatsuchmanifestationsareduetoadeficiencyofthenormalconservativefunctionofthemeibomianlipidinretardingevaporation,andrepresentaninducednon-wettingeyedisease.Analternativeexplanationrelatestobacterialinfectionsofthemeibomianglandsandtosuggestedtoxicmeibomianlipiddegradationproducts.'314Theactionoffattyacidsreleasedbybacteriallipasesisarelatedhypothesis."CasereportThepresentpaperreportsabsenceofmeibomianglandsinayoungcontactlenswearer.InSeptember1981a16-year-oldwhiteAmericangirl(OEH196501)wasreferredtotheOxfordEyeHospital312 CongenitaldeficiencyofmeibomianglandsOBLIQUEGLANDSFig.1Diagramtoillustratethedispositionofresidualmeibomianglands.INCOMPLETEGLANDRUDIMENTSfromanAmericanAirBasewithahistoryofoneandahalfmonthsofsoftcontactlenswearusingcoldchemicalsterilisation.Shereportedincreasingred-nessoftheeyes,andinthetwoweeksbeforepresentationshedevelopedmarkedpaininbotheyes.Shestoppedlenswearforoneweekandwasstartedonchloromycetinsolutionqdswithoutrelief.Amarked'cornealstipple',withpunctatestainingintheleftmorethantherighteye,failedtoresolveoverthisperiod.Acuitywasreducedineacheye.Acuityinth
ecasualtydepartmentwasRE6/9andLE6/12corrected,improvingtoRE6/5andLE6/6+3withapinhole.Therewasbilateralpunctatekeratopathy.Examinationintheexternaldiseaseclinictwodayslatershowedbilateraluppertarsalconjunctivalpapillaeofmilddegreeandafewfolliclesandcysticchangesbelow.Therewasa2+interpalpebralconjunctivalandcornealpunctateepithelialstaining.Anteriorsegmentsandfundiwereotherwisenormal.Atthenextvisititwasrecognisedthattherewasamajorabnormalityofthemeibomianglandswhichwerenotedtobebothdeficientinnumberandabnormalinmorphology(Fig.1).Therightupperlidhad14orificesassociatedwithobliquelydisposedglandsaboutaquartertheirnormallength.Theywereabsentfromthecentralpartofthelid.Inthisregiontherewastheoccasionalsuggestionofaglandwhichfailedtocomeincontactwiththesurfaceofthelidmargin.Therightlowerlidshowedasingleglandandorifice.Onthelefttheupperlidhad16orifices.Glandswereelongatedandobliquelydisposedinthetemporalpartofthetarsalplate,andmoreverticallydisposedinthenasalpartofthelid.Manywereapproximatelyofnormallength.Theleftlowerlidhadasingleshortglandabouthalfthenormallength.Oneithersideofthissolitaryglandwasalineofdark,roundedspotsinthelineofthesubtarsalfold,whichwereassumedtobeabortivetarsalglandrudiments.Therewasnoconnectionwiththelidmargin.Justtemporaltothesolitaryglandorificewasapatchofpresumedsquamousmetaplasiaacrossthebreadthofthelidmargin.Otherwisethelidmarginswerenormalinappear-ance,and,wheremeibomianorificeswerevisible,theyweredisposedattheirnormallocationinthemucocutaneousjunction;slightpoutingwaspresentinsome.Oilwasexpressiblefromtheupperlidglandorificesontheright.Nooilwasexpressiblefromtheotherlids.OvertheperiodDecember1980to1981thefluoresceinbreakuptime(BUT)wasmeasuredonanumberofoccasions:R4-8s,L6-8s;R15s,L5s;R9s,L7s;R5s,L6s.Schirmer'stestwasR15and12mmin5minutes,andL10and5mmonseparateoccasions.Thepatientwasamyope.RefractionwasR-2*75/+0-5axis250=6/4andL-2-75/+050axis170°=6/4.KreadingswereR75along90°;7*35along1500andL75along110°;7*40along300.AboutoneyearafterpresentationshewasrefittedwithaScansoftlensandachievedalldaywear(7.00am-9-00pminFebruary1982).Therewasatendencytostinginginthelefteyeduringwear,afterprolongedstaring.Therewasatendencytoaccumu-latedepositsontherightlens,whichnecessitatedchangingthewearingschedule.DiscussionThepresentstudyreportsapatientwithmeibomianglandswhicharedeficientinnumber,rudimentaryinform,andabnormallydisposed.Theabsenceofanybutminorconjunctivalsigns(interpalpebralstaininganduppertarsalpapillae)andtheyouthofthepatientstronglysuggestthatthisisacongenitalabnormalityoftheglands.Thepatientwasotherwisenormal,andtherewasnoskincomplaint.Therearefewreportsofcongenitalabsenceofmeibomianglands.HollyandLemprefertoanassociationwithanhydroticectodermaldysplasia.16Oneofus(AJB)hasseenasimilarcase,alsofemale,inanotherinstitution.InthispatientRIGHTOBLIQUESHORTENEDGLANDSLEFT313 AJBronandLSMenghersymptomswerepresentfrominfancy.Symptomsinourpatientareassumedtobeduetoadeficiencyofmeibomianoil.Meibomianoilisthoughttopreventevaporationfromtheocularsurface.IntherabbitMishimaandMauriceesti-matedthatmeibomianoilreducedevaporationby4-20fold.""BrownandDervichian'9couldnotconfirmthisconservationaleffectofmeibomianoilonwaterloss.But,althoughhumanmeibomianoilhasadifferentcompositionfromrabbitmeibomianoil,2021itisdifficulttoacceptthatitdoesnotperformthisfunction,sinceitformsamultimolecularsheetacrossthepreocularsurfacebetweenblinksinnormalsubjects.Theotherpostulatedfunctionofmeibomianoilistopreventcontaminationoftheocularsurfacewithsebaceouslipidpresentonthelidskin22andderivedfromlash-associatedsebaceousglands.TheabilityofsebumtolowerthesurfacetensionofpreoculartearmeibomianlipidhasbeengraphicallydemonstratedbyMcDonald,whoshowedthatasebumcoveredhairtouchedontothepreocularsurfacecausesanimmediatedisruptionofthelipidfilm.'ThepresenceofhydrocarbonsinmeibomianoilcollectedbyexpressionfromnormallidS224suggeststhatsomecontaminationofthemeibomiansecretionmayoccurnormally.Nornhasshownthatlipidonthelidmargincanfinditswayintotheconjunctivalsacanddrawsthesameconclusion.Adefic
iencyofnormallidoilinthetearfilmandperhapsthepresenceofsebumonthetearfilmwouldbeexpectedtoencourageevaporationfromtheocularsurfaceandsymptomsandsignsofnon-wetting.TheinterpalpebralstainingpatternandreducedBUTrecordedinourpatientareinkeepingwiththis.InthestudybyBronandTripathi,aparticularformofepithelialsurfacechangewasobservedandtermedcysticepithelialkeratopathy,associatedwiththemeibomianblockadeandotherfeaturesofmeibomianitis."Inthatsituationepithelialcystsareobservedintheexposedregionofthecornea,togetherwithpunctateerosionsandpunctateepithelialkeratopathy.Inourpatientsymptomsappearedafterinstitutionofcontactlenswear,despitethelikelihoodthattheoilglanddeficiencywascongenital.Thissuggestedthatcontactlenswearprecipitatedthesurfacechanges.Punctateinterpalpebralstaininggraduallydecreasedoncecontactlenswearhadbeenstoppedbutdidnotentirelydisappear.Punctatecornealchangesmaybeslowtosubsideaftercontactlensproblemswhatevertheircause;thereforeitisnotcertainthattheirpersistencewasduetothepoorwettingortocontactlens'insult'.Itisourimpressionthatclinicalpresentationofnon-wettingproblemsduetoaqueousdeficiencyormeibomianitisofcon-genitalorearlyonsetdoesnotnecessarilyoccurininfancybutmaystarttoproducesymptomsintheteensortwenties.Thissuggeststhattheocularsurfacemaybemoreresilientintheyoung.Achangeinlensandregimenultimatelypermittedcontactlenswearinthispatient.References1WolffW.Mucocutaneousjunctionoflidmarginanddistributionoftearfluid.TransOphthalmolSocUK1946;66:291-308.2TapieR.EtudebiomicroscopiquedesglandMeibomius.AnnOculist(Paris)1977;210:637-48.3JesterJV,RifeL,NiiD,LuttrullJK,WilsonL,SmithRE.Invivobiomicroscopyandphotographyofmeibomianglandsinarabbitmodelofmeibomianglanddysfunction.InvestOphthalmolVisSci1982;22:660-7.4BronAJ,TiffanyJMT,KauraR,MengherLS.Disordersoftearlipidsandmucousglycoproteins.In:EastyDL,SmolinG,eds.Externaleyedisease.London:Butterworths,1984:63-105.5RobinJB,JesterJV,NobeJ,NicholaidesN,SmithRE.Invivotransilluminationbiomicroscopyandphotographyofmeibomianglanddysfunction.Ophthalmology(Rochester)1985;92:1423-6.6TiffanyJ,DartJ.Normalandabnormalfunctionsofmeibomiansecretion.In:Trevor-RoperP.ed.VIthCongressoftheEuropeanSocietyofOphthalmology.Thecorneainhealthanddisease(1980).RoyalSocietyofMedicineInternationalCongressandSymposiumSeries,No.40.London:RoyalSocietyofMedicine,1981.7ElschnigA.Conjunctivitismeibomiana.MedWochenschr1908;34:1133-5.8FuchsE.Textbookofophthalmology(transDuaneA).Philadelphia:Lippincott,1911:134.9ThygesonP.KimuraSJ.Chronicconjunctivitis.Ophthalmology(Rochester)1963;67:494-503.10KeithCG.Seborrhoeicblepharo-keratoconjunctivitis.TransOphthalmolSocUK1967;87:85-103.11BronAJ,TripathiRC.CysticdisordersofthecornealepitheliumI.Clinicalaspects.BrJOphthalmol1973;57:361-75.12ThygesonP.Bacterialfactorsinchroniccatarrhalconjunctivitis:roleoftoxinformingstaphylococci.ArchOphthalmol1937;18:373-87.13McCulleyJP,SciallisGF.Meibomiankeratoconjunctivitis.AmJOphthalmol1977;84:788-93.14AllenJH.Staphylococcalconjunctivitis.AmJOphthalmol1977;20:1025-31.15McCulleyJP,DoughertyJM.Bacterialaspectsofchronicblepharitis.TransOphthalmolSocUKinpress.16HollyFJ,LempMA.Tearphysiologyanddryeyes.SurvOphthalmol1977;22:69-87.17MishimaS,MauriceDM.Theoilylayerofthetearfilminevaporationfromthecornealsurface.ExpEyRes1961;1:39-45.18MishimaS,MauriceDM.Theeffectofnormalevaporationontheeye.ExpEyeRes1961;1:46-52.19BrownSI,DervichianDG.Theoilsofthemeibomiangland.ArchOphthalmol1969;82:537-49.20TiffanyJM.ThemeibomianlipidsoftherabbitI.Overallcomposition.ExpEyeRes1979;29:195-202.21NicolaidesN,KatarantaJK,Rawdah,TN,MacyJI,BoswellFMIII,SmithRE.Meibomianglandstudies:compositionofsteerandhumanlipids.InvestOphthalmolVisSci1981;20:522-36.22HollyFJ.Tearfilmphysiology.AmJOptomPhysiolOpt1980;57:252-7.23McDonaldJE.Surfacephenomenaoftearfilms.TransAmOphthalmolSoc1968;66:905-39.24TiffanyJM.Individualvariationsinhumanmeibomianglandlipidcomposition.ExpEyeRes1978;27:289-300.25NornMS.Naturalfatsinexternaleye.VitalstainedbySudanIIIpowder.ActaOphthalmol(Kbh)1980;58:331-6.Acceptedforpublication30May1986.31