Total thyroidectomy in HIV positive patient with buffalo hump and taurine neck

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InternationalJournalofSurgeryCaseReports61(2019)64–66

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International

Journal

of

Surgery

Case

Reports

jo u r n al ho me p a g e :w w w . c a s e r e p o r t s . c o m

Total

thyroidectomy

in

HIV

positive

patient

with

buffalo

hump

and

taurine

neck

Elena

Carrese,

Uliano

Morandi,

Alessandro

Stefani,

Beatrice

Aramini

∗

DivisionofThoracicSurgery,DepartmentofMedicalandSurgicalSciencesforChildrenandAdults,UniversityHospitalofModena,ViaLargodelPozzon. 71-41124Modena,Italy

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received12May2019 Accepted11July2019 Availableonline19July2019

Keywords: Thyroidectomy Lipodystrophy

Highlyactiveanti-retroviraltherapy (HAART)

HIVpositive

a

b

s

t

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t

BACKGROUND:TheauthorspresentacaseofmultinodulargoiterinanHIV-positivepatientaffectedby lipodystrophywithparticularaccumulationofadiposetissueintheregionoftheneckandtrunk. CASEPRESENTATION:Thepatient,a53-year-oldman,presentedwithmultinodularstrumawithpartial retrosternalengagement,aswellasmultiplethyroidnodulesincreasinginsize;someofthenoduleshad suspiciouscharacteristicsonultrasound.Needleaspirationbiopsywasdifﬁculttousetodeterminethe presenceoflipodystrophy;however,evenintheabsenceofcytology,surgicaltreatmentwasnecessary duetothepresenceofdyspneaduringexercise,thedimensionofthegoiterwithretrosternal engage-ment,andtheovalizationofthetracheallumen.Thepatientunderwenttotalthyroidectomybyanterior cervicotomywithparticularattentiontopatientpositioningbecauseofthebuffalohumpandtaurine neck.Histologicalexaminationwaspositiveforadenomatoushyperplasiawithoutbreaksofpapillary microcarcinoma.

CONCLUSIONS:Theaimofthiscasereportwastohighlighttheimportanceoftheperioperativeteamwork, withparticularattentiontopatientpositioningbeforesurgery,aswellasprofessionalcollaborationand experienceamongtheoperators.

1. Background

Highlyactiveanti-retroviraltherapy(HAART)withassociated lipodystrophysyndromereferstoaseriousmetabolicsyndromein HIV-infectedpatientsreceivinghighlyantiretroviraltherapy[4].It isgenerallycharacterizedbymetabolicchangessuchasthe devel-opment of dyslipidemia, insulin resistance, glucose intolerance and abnormal redistribution of body fat (peripheral lipoatro-phyandlipohypertrophy)[3].Lipohypertrophyreferstolocalized abnormal fat accumulation and most commonly occurs in the intra-abdominal compartment (visceral adipose tissue), breast, dorso-cervicalarea(buffalohump)ordiscretelyaccumulatesunder theskin(lipomas).Lipoatrophyischaracterizedbylossof

subcu-Abbreviations: HIV, human immunodeﬁciencyvirus; HAART,highlyactive anti-retroviraltherapy;AZT,zidovudine;Ddi,didanosine;d4T,stavudine;3TC, lamivudine;IDV,indinavir;EFV,efavirenz;BMI,bodymassindex;HCV, hepati-tiscvirus;ASA,Americansocietyofanesthesiologists;PIs,proteaseinhibitors; NRTIs,nucleosidereversetranscriptaseinhibitors;LDL,low-densitylipoprotein; HDL,high-densitylipoprotein;non-NRTIs,non-nucleosidereversetranscriptase inhibitors.

∗ Correspondingauthor.

E-mailaddresses:[email protected](E.Carrese),

[email protected](U.Morandi),[email protected]

(A.Stefani),[email protected](B.Aramini).

taneousfat, particularlyin theface,buttocks, armand leg.The coexistenceoflipohypertrophyandlipoatrophyisreferredtoas mixedlipodystrophy[5].Thissyndromewasidentiﬁedin1998, butitscausesarenotfullyunderstood;currentdatasuggesta mul-tifactorialpathogenesis,withthemajorcontributingfactorsbeing thechoiceoftreatment,durationoftreatmentandpatient-related factors,suchasage,BMI,andthelevelofimmunodeﬁciency[3,5,4]. Theaimofthisreportwastoshowperioperativemanagement inanHIVpatientaffectedbylipodystrophysyndromewho under-wentthyroidectomyforamultinodulargoiter.Theworkhasbeen reportedinlinewithSCAREcriteriahasbeenreportedinlinewith theSCAREcriteria[6].

2. Casepresentation

We present a case of a 53-year-old man who came to our departmentfor alargemultinodulargoiterwithmultiple suspi-cious nodules onultrasound. The diagnosis wasmade in 1999 duringhospitalizationforanepisodeofthyrotoxicosis.Thepatient wasdiagnosedasHIV-positivein1994andbegan antiretroviral treatment with AZT (zidovudine) and Ddi (didanosine) with minimal initial beneﬁt;however, subsequent worsening of the immunological condition was noted. Therefore, beginning in June 1997, therapy was switched to d4T (stavudine) plus 3TC (lamivudine) and IDV (indinavir); this achieved excellent viral

https://doi.org/10.1016/j.ijscr.2019.07.020

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E.Carreseetal./InternationalJournalofSurgeryCaseReports61(2019)64–66 65

Fig.1. A-C-Dshowalateralviewofthepatientaffectedbylipodystrophywithparticularaccumulationofadiposetissueintheregionoftheneckandtrunk.Fig.2Bshowsa posteriorview.

replicationcontrolandimprovedtheimmunologicalcondition.In 1999,achangeinHIVtreatmentwasnecessaryduetothepresence ofanimportantlipiddysmetabolismwithhypertriglyceridemia, modiﬁcationof thehabitus withan importantlipohypertrophy ofthecervicodorsalregion(buffalohumpandtaurineneck)and lossofsubcutaneousfatofthelegs(Fig.1A–D).TherapywithAZT (zidovudine)+3TC(lamivudine)+EFV(efavirenz)enabledcontrol of viral replication and stability of the lipodystrophic picture. Thepatienthadundergonemultipleliposuctions,buttheywere noteffective,likelyduetotheexcessiveaccumulationofadipose tissue.Inouropinion,themostrelevantclinicalaspecttoconsider wasthelargebuffalohumpandtaurineneckduetolipodystrophy. He also presented with negative thyroid autoimmunity, with normal calcitonin and calcemic blood values. The ultrasound examshowedstrumawithpartialretrosternalengagementand multiplethyroidnodulesincreasinginsizerelativetoaprevious check,as wellas suspicious features for neoplasia. The patient hadundergoneseveralneedleaspirations,whichfailedduetothe thicknessandabundancyoftheadiposetissue.

Patientcomorbiditiesweremainlybasedonthealterationof glucosemetabolism(duetodiabetesmellitus),alterationofbone metabolism(duetoosteopenia),severeobesity(bodymassindex, BMI,of34.6),andhypertension,aswellasslightlimitationin run-ning, weightliftingand playingintense sports.Healsoreported thatapreviousHCVhepatopathyhadbeentreatedwithHarvoni. Theindicationtoremovethethyroidglandswassuggestedbythe patient’sendocrinologist,primarilyduetothesuspiciousnodules. PreoperativeanesthesiaevaluationpredictedanAmericanSociety ofAnesthesiologists(ASA)anestheticriskof3,requiringcomplex

Fig.2.Chestx-rayshowstheovalizationofthetracheallumen(arrow).

intubationforthepatient’s habitusandfortheshrinkageofthe trachea.Indeed,preoperativechestX-rayshowedanovalizationof thetracheallumen(11mm)toaplanepassingthroughC7(Fig.2). Aftercarefulpositioning(Fig.3AandB),thepatientunderwent totalthyroidectomybyanteriorcervicotomy.Totalthyroidectomy wasperformedwithtechnicaldifficulties:first,intubationofthe patientwasrequiredfortheoperationduetothehyperextension oftheneck;second,accesstothegoiterwasdifficultduetothe amountofadiposetissue,aswellasthebuffalo humpand tau-rine neck. Thyroidisolation wascomplicated on the rightside of the gland for the increasedvolume, particularly due to dif-ficulty in recurring laryngeal nerve isolation. No intraoperative complicationsandnobleedinglosswereobservedduringorafter surgery.Thedrainswereremovedonthesecondpostoperativeday. Despitethepatient’sHIVinfection,nocomplicationswerenoted aftersurgery,especiallyaroundthewound.Calciumbloodvalues stayedinthenormalrange aftersurgery.Thepatientpresented

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66 E.Carreseetal./InternationalJournalofSurgeryCaseReports61(2019)64–66

withdysphonia; therefore, direct laryngoscopy wasperformed, whichshowedﬁxationoftherightvocalstringwithgood phona-torycompensation,aswellasconsistencyoftherespiratoryspace. Therefore,thepatientwassent tologopedicrehabilitationafter dischargefromthehospital.

3. Discussionandconclusion

Lipodystrophy syndrome is an important complication of antiretroviraltherapyduetoitsincreasedcardiovascularriskasa resultofmetabolicalterations,aswellasthepsychologicalaspects andqualityoflife.

Studieshave shown that protease inhibitors (PIs)and some nucleoside reverse transcriptase inhibitors (NRTIs), especially stavudine(d4T),increasethelevelsoftriglycerides,total choles-terolandlow-densitylipoproteincholesterol(LDL);however,they reduce high-density lipoprotein (HDL) levels. In contrast, regi-menswithnevirapineandefavirenz(non-NRTIs,orNNRTIs)show reducedatherogeniceffectsonlipidprofiles[3,1].PIsandNRTIs appear to cause mitochondrial toxicity, modifying the enzyme activity involved in metabolism [7,8]. Other studies support a directroleforHIV:adiposetissuemayserveasreservoirforHIV, altering the local tissue environment and promotingenhanced adiposetissueinflammation.HIVproteinsalteradiposetissue func-tionandincreaseinflammation;therefore,HIV-associatedchronic inflammationandimmuneactivationmayplayadirectroleinthe developmentoflipohypertrophy[5].

Theriskofdevelopinglipodystrophyisgreaterwithadvanced age,inpeoplewhohavethelongestdurationsofinfectionandin patientswhohavehadtherapyforlongerperiods[3,4]. Further-more,somestudieshaveshownthatpatientswithlipoatrophyhave signiﬁcantlylowerBMIthanthosewithlipohypertrophy[2,8].

The combination of obesity, lipodystrophy, central adipos-ity,dyslipidemia,andinsulinresistancecommonlyoccursamong HIV-infectedadults;theseconstituteriskfactorsassociatedwith cardiovasculardiseases,whichrepresentthethirdmostcommon causeofdeathamongHIV-positiveindividualsinEurope. More-over,lipodystrophicbodychangescaninﬂuencethequalityoflife, leadingtolowadherencetoHAARTandsubsequentvirologicand clinicalfailure[3,9,4,5].

Inthiscase,lipodystrophysyndromewasacomplicated obsta-cletothenecessaryintervention.Surgery,despiteallofitstechnical difﬁculties,wastheonlymethodtoobtainthediagnosis consider-ingthefailedaspirationattempts.Moreover,thepatienthadshown progressionofthyrotoxicosis,whichjustiﬁedthechoiceof surgi-calintervention.Thesurgicalindicationwascorrectbecausethe histologicalexaminationwaspositiveforpapillarycarcinoma.

Inconclusion,webelievethateverymedicalorsurgical inter-ventionmustbeaimedatimprovingormaintainingthepatient’s health and quality of life; therefore, we must trytoovercome technicalcomplexitywherepossible.Thecollaborationofan expe-riencedteamisessentialtoobtainsatisfactoryresults,especiallyin themostchallengingcases.

DeclarationofCompetingInterest

Theauthorshavenoconﬂictsofinteresttodisclose. Funding

Nofunding

Ethicsapproval

ForsinglecasereportNOethicalapprovalneeds.Patientsigned aconsentforpublishingthecasereport.

Consent

Patientsignedaconsentforthepublicationofthiscasereport. Authorcontribution

ECandBAwrotethecasereport.ASandUMrevisedthecase report.

RegistrationofResearchStudies

EthicalBoardapprovalisnotrequiredforcasereportsinour Center.

Guarantor

Prof.UlianoMorandiistheGuarantorofthiscasereport. Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed Acknowledgements

Notapplicable. References

[1]E.Jones,P.Mazirka,M.A.McNurlan,F.Darras,M.C.Gelato,G.Caso,Highly activeantiretroviraltherapydysregulatesproliferationanddifferentiationof humanpre-adipocytes,WorldJ.Virol.6(3)(2017)53–58,http://dx.doi.org/10.

5501/wjv.v6.i3.53,Epub2017Aug12.

[2]N.S.Kumar,J.Shashibhushan,VenugopalK.Malappa,H.Vishwanatha,Menon M.J.Clin,LipodystrophyinHumanImmunodeﬁciencyVirus(HIV)Patientson HighlyActiveAntiretroviralTherapy(HAART),DiagnRes.9(July7)(2015) OC05–8,http://dx.doi.org/10.7860/JCDR/2015/12979.6183,Epub2015Jul1.

[3]M.G.Ceccato,P.F.Bonolo,A.I.SouzaNeto,F.S.Araújo,M.I.Freitas,Antiretroviral

therapy-associateddyslipidemiainpatientsfromareferencecenterinBrazil,

Braz.J.Med.Biol.Res.44(November11)(2011),1177-83.Epub2011Oct7.

[4]A.Balasubramanyam,R.V.Sekhar,F.Jahoor,P.H.Jones,H.J.Pownall,

PathophysiologyofdyslipidemiaandincreasedcardiovascularriskinHIV

lipodystrophy:amodelof’systemicsteatosis’,Curr.Opin.Lipidol.15(February

1)(2004)59–67,Review.

[5]J.E.Lake,T.L.Stanley,C.M.Apovian,S.Bhasin,T.T.Brown,J.Capeau,J.S.Currier, M.P.Dube,J.Falutz,S.K.Grinspoon,G.Guaraldi,E.Martinez,G.A.McComsey, F.R.Sattler,K.M.Erlandson,Practicalreviewofrecognitionandmanagementof obesityandlipohypertrophyinhumanimmunodeﬁciencyvirusinfection,Clin. Infect.Dis.64(May10)(2017)1422–1429,http://dx.doi.org/10.1093/cid/

cix178,Review.

[6]R.A.Agha,M.R.Borrelli,R.Farwana,K.Koshy,A.Fowler,D.P.Orgill,Forthe

SCAREGroup,TheSCARE2018statement:updatingconsensussurgicalCAse

REport(SCARE)guidelines,Int.J.Surg.60(2018)132–136.

[7]S.Esser,D.Helbig,U.Hillen,J.Dissemond,S.Grabbe,SideeffectsofHIV therapy,J.Dermatol.Ges.5(September9)(2007),http://dx.doi.org/10.1111/j.

1610-0387.2007.06322.x,745-54.Review.

[8]C.Dell’Isola,L.Aprea,T.Pizzella,C.Izzo,Effectofanti-retroviraltherapyon

bodycompositionchanges:aliteraturereview,Infez.Med.14(September1)

(2006)5–12.

[9]J.Falutz,Therapyinsight:body-shapechangesandmetaboliccomplications associatedwithHIVandhighlyactiveantiretroviraltherapy,Nat.Clin.Pract. Endocrinol.Metab.3(September9)(2007)651–661,http://dx.doi.org/10.

1038/ncpendmet0587.

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