RevBrasAnestesiol.2014;64(2):124---127
REVISTA
BRASILEIRA
DE
ANESTESIOLOGIA
OfficialPublicationoftheBrazilianSocietyofAnesthesiologywww.sba.com.br
CLINICAL
INFORMATION
Transient
unilateral
combined
paresis
of
the
hypoglossal
nerve
and
lingual
nerve
following
intubation
anesthesia
Hulya
Ulusoy
∗,
Ahmet
Besir,
Bahanur
Cekic,
Muge
Kosucu,
Sukran
Geze
DepartmentofAnesthesiologyandCriticalCare,FacultyofMedicine,KaradenizTechnicalUniversity,Trabzon,Turkey
Received8November2012;accepted14December2012
Availableonline11October2013
KEYWORDS
Hypoglossalparalysis; Lingualparalysis; Intubationanesthesia
Abstract Nervedamagemayoccurinthepharyngolaryngealregionduringgeneral anesthe-sia.Themostfrequentlyinjurednervesarethehypoglossal,lingualandrecurrentlaryngeal. Theseinjuriesmayariseinassociationwithseveralfactors,suchaslaryngoscopy,endotracheal intubationandtube insertion,cuffpressure, maskventilation, thetriple airwaymaneuver,
theoropharyngealairway,manner ofintubationtube insertion,headandneckpositionand
aspiration.
Nerveinjuries inthisregion may take theform ofanisolated singlenerve orofparesis oftwonervestogetherintheformofhypoglossalandrecurrentlaryngealnervepalsy(Tapia’s syndrome).However,combinedinjuryofthelingualandhypoglossalnervesfollowingintubation anesthesiaisamuchrarercondition.Theriskofthisdamagecanbereducedwithprecautionary measures.Wedescribeacaseofcombinedunilateralnervushypoglossusandnervuslingualis paresisdevelopingafterintubationanesthesia.
©2013SociedadeBrasileiradeAnestesiologia.PublishedbyElsevier EditoraLtda.Allrights reserved.
Background
and
objectives
Withintheframeworkof generalanesthesia(GA), postop-erativeairwaycomplicationsdonothaveanegativeimpact onpatient comfort. Unilateral or bilateral nerve damage occupiesanimportantplaceamongthesecomplications.1---3
Themostcommonnervedamage-associatedcomplications arerecurrentlaryngealnerve(RLN)palsy,hypoglossalnerve
∗Correspondingauthor.
E-mail:hulyaulusoy.md@gmail.com(H.Ulusoy).
palsy,andpalsyoftheexternalbranchofthelaryngealnerve orlingualnerve.Hanschetal.determineda1.9%incidence ofunilateral RLNpalsyduringlaryngoscopy.3 Thenet
inci-dence associatedwithisolatedhypoglossus nervepalsyor lingualnervepalsyisunknown,althoughitislow.4---6Cases
of isolated hypoglossal nerve palsyor lingual nerve palsy associatedwithendotrachealintubationhavebeenreported aftertheuseofthelaryngealmask airway(LMA)inrecent years.2,7---9Wediscussacaseofcombinedunilateral nervus
hypoglossus and nervus lingualis paresis developing after septorhinoplasty surgery under GA in the light of the lit-erature.
0104-0014/$–seefrontmatter©2013SociedadeBrasileiradeAnestesiologia.PublishedbyElsevierEditoraLtda.Allrightsreserved.
Transientunilateralhypoglossalnerveandlingualnervepalsy 125
Case
report
A 19-year-old female patient in the ASA I risk group was operated by an ENTsurgeondue tonasoseptaldeformity. The patienthad been operated on 2 years previously for the same reason but had experienced no GA-associated problem.Preoperativeexaminationrevealeda4-cmmouth opening, and the soft palate and uvula were visualized. HerMallampatiscorewas2,andthethyromentaldistance andsternomental distance were normal.Atlanto-occipital joint extension was adequate. GA was performed using intravenous(iv)pentothal(5mg/kg),fentanyl(1g/kg)and vecuronium (0.1mg/kg). Mask ventilation was performed withdifficultyusingabilateraltriplemaneuverinorderto reduceairleakageduetoface-maskincompatibility.ANo. 4airwaywasinsertedtoimproveventilation.Laryngoscopy wasperformedusingaMacintoshblade(No.3).Cricoid pres-surewasappliedtoimprovevisualization.Anendotracheal tube(ETT)(7.5mm,redrubber;RuschTM)wasinsertedinto
thetracheaatthefirstattempt.Oncetheplacementofthe tubehadbeenconfirmed,thecuffoftheETTwasinflated soasnottoexceed20cmH2O.ThelocationoftheETTinside
themouthwasalteredtotheleftsideofthetonguedueto thepositionofthe patientanesthesiadevice. Itwaslater located by attachment to the edge of the left lowerlip. GAwasmaintainedwithasevofluraneandoxygen/N2O
mix-ture.Theoperationwascontinuedwiththeheadandneck inthecentralline,withthepatientinasemi-supineposition withslightextensiontotheanterior.Asmallthroatpackwas insertedalongsidethetubeandremovedbeforeextubation. Surgery lasted180min, andthe patient’s vital findings were stable. The patient was extubated with no prob-lem and monitoredin the PACUuntil attaininga Modified AldreteScore≥9.Sorethroatandhoarsenessappeared dur-ingobservationonthewardonthe1stdaypostoperatively. Examinationrevealedapalpablemass,approximately1cm in diameter, in the angulus mandibulae in the left sub-mandibular region. At indirect laringoscopicexamination, theuvulawasedemicandhyperemicandvocalcord move-mentswerefree.The patientwastold thatthecondition mightbetemporary,andanti-inflammatorytherapywas ini-tiated.Difficultyinswallowingandspeakingandinabilityto usethetonguedevelopedonthe2ndday,andneurological andENTexaminationswereperformed.Noabnormalitywas determinedatexternaloralexamination,thoughtherewas pronouncededemainthetongue,particularlyontheleft, restrictedtonguemovementtotherightanddeviationtothe left(Fig.1).Onthe3rddaypostoperatively,pronouncedloss of taste sensationwasdetermined inthe fronttwo-thirds ontheleftofthetongue.Othercranialnerveexaminations werenormal.Apartfroma1-cmlymphadenopathyintheleft submandibularregion,noadditionalpathologywas identi-fied at MRI of the head and neck. Unilateral paralysis of thenervushypoglossusandnervuslingualiswasdiagnosed. The patientwas explained that this nerve damage might alsoinvolvepermanentsymptomsandthetreatment proto-colwassetout.Intravenousprednolatadosageof1mg/kg wasusedforthefirst3days,followedbyperoral adminis-tration.Speechandswallowingtherapywereinitiated.The dosagewasgraduallyreducedafterthe10thday,andsteroid wasstoppedonthe 20thday. Vitamins E,BI and B6were continuedfor1month. However,whennervedamagewas
Figure1 Deviationtotheleftinthetongueassociatedwith
unilateral paralysis of the hypoglossal nerve on the 3rd day
postoperatively.
diagnosedthepatientexperiencedsevereanxietyandsleep impairment,andaconsultationwasheldwiththe psychia-trydepartment.Thepatientexhibiteddepressiveaffectivity andanxietyatevaluation andwasdiagnosedwith depres-sion,anxietydisorderandconversiondisorder.Theselective serotoninreuptakeinhibitor(SSRI)essitalopram(CIPRALEX®
FilmTablet,Lundbeck,Denmark)wasstartedatadosageof 10mgpoonthe4th day.There wasaslightimprovement insore throatand swallowingdifficultyonthe 7thday of treatment.Pronouncedatrophywasseenintheleftlingual mucosa.Speechimpairment,inabilitytousethetongueand lossofsenseoftastebegantodeclineatexaminationsinthe 3rdweek.Therewasasignificantimprovementintaste per-ceptionandtheatrophicappearance inthetongue atthe end of the 3rd month. The patient’s anxiety, depression, sleeplessnessand problems associatedwithisolation from societyimprovedandSSRItherapy wasstoppedattheend ofthe12thweek.Allsymptomsresolvedbytheendofthe 6thmonth.
Discussion
Awide rangeof upper airway problems maybe observed post-GA, from minor sore throat and pharyngeal dryness toimpaired speech,hoarsenessandrespiratorydifficulty.1
In the case described here, combinedneuropraxia of the hypoglossalandlingualnervesdevelopedfollowing intuba-tionGAforseptorhinoplastysurgery.
Isolatedhypoglossalnervedamageassociatedwith laryn-goscopy and tracheal intubation is usually unilateral and is seen as a rare postoperative complication with multi-factorial causes.2,10 Other causes includeuse of the LMA,
application of cricoid pressure and direct compression of thehypoglossal nervebeneath the angle of themandible angleduringmaskventilation.11,12Itisnotablethatinmost
case reports unilateral hypoglossal nerve injury develops onthe left side.8,11,12 Hypoglossal paresis may sometimes
not be isolated after endotracheal intubation, and may becomorbid withvocalcordparalysis(RLN palsy)(Tapia’s syndrome).4Mostofthesecasereportshaveemergedafter
126 H.Ulusoyetal.
andneckarenerveinjuryrisksassociatedwiththesurgery. Largethroatpacksinparticularcancausedirectlingualand hypoglossalnerveinjuryinthisoperation.7,13---15
Lingualnervedamageismorecommonlyseenduring den-talprocedures.Itisrarerin associationwithlaryngoscopy andendotrachealintubation.16Silvaetal.reportedlingual
numbnessandlossoftastesensation24hafterconclusionof anesthesiainanobesepatientwhocouldnotbeintubated.7
However,lingualnervedamagemayalsoappearinthe48th to72ndhoursaswellasimmediatelyafteranesthesiaand recovery.7,9
Evers et al. described a case of combined hypoglos-sal and lingual nerve palsy with thickening of the soft tissue in the tongue and laryngeal structures in asso-ciation with acromegaly and showed that prognathism facilitateslaryngoscopy-associatedlingualnerveinjury.6In
thatacromegalicpatient,hyperextensionoftheheadduring transsphenoidalhypophysectomyor thetube beingtightly attached in the neck may have caused combined injury ofthehypoglossalandlingualnerves.6 TeichnerandJones
reportedthatpressuretothecricoidcausedhypoesthesiain thetongueintwoseparatecases.17,18 Thecricoidpressure
maneuvermaycausestretchingatthepointwherethe lin-gualnervepassesthroughthehypoglossus muscle.Onthe otherhand,Wangshowedthatimproperplacementofthe oropharyngealairwaymayleadtolingualnervedamage.19
James revealed that the lingual nerve can be injured by anteriordisplacementofthemandible.20
There are very few cases of combined nerve damage intheliterature,andtheanatomicalcoursesofthesetwo nervesneedtobeexaminedinordertoaccountfor concomi-tantinjurytothem,asinourcase.4---6Thehypoglossalnerve
runsbeneaththesubmandibulargland,thesubmandibular ductandthelingualnerveandintersectsintheanteriorof thehypoglossusmuscle.Thisiswherethelingualmuscleis mostsuperficial. Cricoid pressure,the triple maneuver or airwayusecanallbecomeriskfactorsinthissituation.
We evaluated the severity of the symptoms and the lengthoftimetorecoveryfromthenerveinjuryinourcase as‘‘neuropraxia’’(classI;physiologicalblock)accordingto the Seddon classification (1943) and ‘‘type 1’’ according totheSunderland classification(1951).Theformofnerve injurywasinallprobabilitycompressiontype,similartothat intheliterature.16
Inouropinion,theremaybefive(other)possiblecauses, in addition to laryngoscopy, of the combined unilateral nervushypoglossusandnervuslingualisparalysisinthiscase report. First, mask compression and the triple maneuver wereperformedtoensureadequateventilationduring anes-thesiainduction(constantbilateralanteriorpressureatthe mandibularangles).Second,theoropharyngealairwaywas intheoralcavityduringthetriplemaneuver.This maneu-vermay haveincreasedthe pressureeffectof theairway on the tongue. Third, cricoid pressure was applied dur-inglaryngoscopyinordertoimprovevisualization.Fourth, theETTusedwashigh pressure,low volume,non-flexible red-rubber, and the ETT was relocated from the right to the left non-flexible. Fifth, although the head and neck werenotplaced inan extreme anteriorposition,theETT wastightlyattached.Althoughtheseprobablyfactorsmay appearharmlessinday-to-daypractice,theirsimultaneous occurrencemayhavefacilitatednerveinjuries.
Thereis noprovenbeneficialprotocolforcranialnerve injuriesintheliterature,althoughthebenefitsofhigh-dose steroid therapy have been shown in Bell’s palsy.21
Corti-costeroidsmayreducesecondary injurybyreducingtissue edema andinflammation.16 Inadditiontosteroidtherapy,
combinationsofanti-inflammatorydrugsandvitaminBare alsoagreedtobebeneficial.However,uncertaintyoverthe outcomes of therapy and impaired speech and sense of taste, significant components of daily life, may also give rise tosevere psychiatricproblems. Individual social fear andanxietyrequirepsychiatricmonitoring andtreatment. Psychiatricsymptommaydevelop inparallelto neurologi-calsymptoms. Neuropraxia-typeinjuries generallyresolve in4---6monthswiththistherapy,asinourcase.
Conclusion
Combined paralysis of the hypoglossal and lingual nerves followingorotrachealintubationforGAis ararecondition in the literature. Importance must be attached to early postoperativevisitsandcarefulquestioningofthepatient, and to collaboration between the neurologist, otolaryn-gologist and, if required, the psychiatrist. Accurate and carefulairwaymanagementandroutineproceduresduring GAcanreducetherisksduringanesthesiaandextubation.In ordertoreducetheiranxiety,patientsshouldbeadequately informedabouttheresolutionofgenerallytemporary symp-toms.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
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