Primary and isolated thyroid Hodgkin's lymphoma: A case report

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CASE REPORT – OPEN ACCESS

InternationalJournalofSurgeryCaseReports37(2017)134–138

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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

Primary and isolated thyroid Hodgkin’s lymphoma: A case report

Mohammed Moutaa Tatari

^∗

, Said Anajar, Sami Rouadi, Reda Abada, Mohammed Roubal, Mohammed Mahtar

ENTDepartment,FaceandNeckSurgery,HospitalAugust,20’1953,UniversityHospitalCentreIBNROCHD,Casablanca,Morocco

a r t i c l e i n f o

Articlehistory:

Received11January2017

Receivedinrevisedform1May2017 Accepted1May2017

Availableonline25May2017

Keywords:

Thyroid Hodgkin’s Lymphoma Nodularsclerosis Casereport

a b s t r a c t

INTRODUCTION:Hodgkin’slymphomararelyinvolvesthethyroidgland.Itistypicallypresentedasafast growingneckmassthatissometimesaccompaniedbyrespiratorycompressionsymptoms.

CASEREPORT:Wereportoneofthefew(theseventeenth)caseofprimaryandisolatedHodgkin’sthyroid lymphomapresentedbya65yearsoldman,consultingforafastgrowingneckmasswithHodgkin’s symptoms.Thepatienthadtotalthyroidectomyandshortcoursesofchemotherapy,thentotalresolution ofsymptomatology.

CONCLUSION:MostthyroidHodgkin’slymphomaarepresentedbywomen,rarelyman,isolatedand primary.Since1962,weonlyfoundsixteencasesdescribedintheliterature.Hodgkin’slymphomashould beconsideredinthedifferentialdiagnosisofpatientswithathyroidmassforrapidmanagement.

1. Introduction

Primarylymphomaofthethyroidglandisararetumour,withno clinicalandparaclinicalspeciﬁcities,accountingforonly5%ofthy- roidmalignanciesand2%ofextranodallymphomas[1].Hodgkin’s lymphomararelyinvolvesthethyroidgland.

Thediagnosisishistological.Treatmentisbasedonchemother- apy,monoclonal antibody and radiotherapy. The surgery must beavoidedwhenthediagnosiscanbeobtainedbeforeorduring theintervention, butthyroidectomymustbedoneandit isthe mainwaytogethealinginassociationwithchemotherapywith orwithoutradiotherapy.OnlyrarecasesofHodgkin’slymphoma presentinginthethyroidhavebeenreportedintheliterature[2].

Wereport thecaseof a 65years old man consultinginour ENTdepartment,20August1953Hospital,CasablancaMoroccofor primaryandisolatedthyroidHodgkin’slymphoma.

ThisworkhasbeenwritteninaccordancewiththeSCAREcrite- ria[3].

2. Casereport

Wereportthecaseofa(Itisabout)65yearsoldman,livingin Casablanca,Morocco,withnomedicalorsurgicalhistory,admitted inourENTdepartmentforananteriorandmedialcervicaltume-

∗ Correspondingauthorat:ENTDepartment,FaceandNeckSurgery,Hospital August,20’1953,UniversityHospitalCentreIBNROCHD,streetaitbaha,bdBordeaux N5,Casablanca,Morocco.

E-mailaddresses:tatarimoutaa@hotmail.com(M.M.Tatari), anajar.said.med@gmail.com(S.Anajar).

faction,whichstartedgrowing8monthsago,rapidlyincreasing involumewithoutpain.Theothersymptomsweregeneralpru- ritus,nightsweatsandfatigue,withnofever,noemaciation,no dyspnea,nodysphoniaorthyroidglanddisorder.Therewereno similarcasesreportedinthepatient’sfamily.Thepalpationfounda hardtumefation,andnopalpablecervicallymphnodes.Thegeneral physicalexaminationdidn’tfindanyhepatomegaly,orsplenome- galiaor otherclinically palpablelymphnodsin thebody.Blood countcellsshowedadisorderoflymphocytesthatwereslightly increased.Acceleratedsedimentationrate.CervicalandthoracicCT scansweredoneshowingatissuemassoftherightthyroidlobedip- pingtotheanteriorandmiddlemediastinum.Thyroidfineneedle aspirationwasperformedbeforethyroidectomy.Itcontainedsome atypicalcells,raisingthepossibilityofHodgkin’slymphoma.Atotal thyroidectomywasdecided.Thepatientwasoperatedwithoutinci- dent,witha goodpostoperativewarning,withoutdysphoniaor dyspneabytraumaofthelaryngealrecurrentnerves.Thepatientis underLevothyroxinesodium100␮gperday.Allthesymptomsof thepatienthavecompletelydisappearedafterthyroidectomy.The histologicalstudyshowedascleronodularHodgkin’slymphoma confirmedbytheimmunohistochemicalstudywhichbringouta stronganddiffusepositivityofthetumorcellstotheanti-CD-15 andanti-CD20antibodies.ItisclassifiedIB(Ifortheinvolvementof asinglelymphnoderegion,sothyroidgland,andBforthepresence ofsystemicsymptoms).

Thepatient wasrefered tothe haematologydepartmentfor furthertreatments.Thepostoperativecoursewasuneventfuland thepatientbeganchemotherapytreatmentincludingfourcycles ofcombinedcyclophosphamide,doxorubicin,prednisoneandvin- cristine.Finally,surgeryandchemotherapyrealizedthestablecure

http://dx.doi.org/10.1016/j.ijscr.2017.05.007

creativecommons.org/licenses/by-nc-nd/4.0/).

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M.M.Tatarietal./InternationalJournalofSurgeryCaseReports37(2017)134–138 135

Fig.1. XXX.

Fig.2. XXX.

ofthediseaseandthepatientisaliveaftertwoyearswithoutrecur- renceormetastases(Figs.1and2).

TheABVD(Dacarbazine,Bleomycin,Vinblastineand Doxoru- bicin) protocol was established, four courses were done. PET scan,cervicothoracoabdominopelvicscaniscompletelynormalin postoperatory,andthebloodcountisbalancedwithanormallym- phocyterate.

3. Discussion

Thyroidcarcinomaisthemostcommonendocrinemalignancy howeverprimarythyroidlymphoma(PTL)accountsfromonly1–5%

ofallthyroid malignancies.B-celltypenon-Hodgkinlymphoma (NHL)isafrequentlydescribedtypeofPTL,whileHodgkin’sand T-celllymphomaarerare[1].Thyroidlymphomatypicallypresents witharapidlyenlargingneckmassleadingtocompressivesymp- toms[4].However,primarythyroidlymphomadevelopsinonly 0.5%ofallcasesofHashimoto’sthyroiditis[5].Duetothisunder- lyingriskfactor,primarythyroidlymphomatypicallyoccursmore ofteninwomenthanmen(8:1)andusuallylaterinlife(sixthor seventhdecade)[6].

Inthisstudy,itisaboutaman,whichismakingthecasemore interestingandrare.

Andthis patient presentscleronodular Hodgkin’slymphoma anditisrarelydescribedintheliterature.

ThelesionhavinganeventualhistoryofHashimoto’sthyroiditis (HT)appearsasa moreorless rapidlyenlarginganteriorcervi-

calmassassociatedornotwithlymphadenopathywhichintime addsymptomsrelatedtocompressionsuchashoarseness,dysp- neaanddysphagia.Inourcase,noknownantecedentofthyroidite.

Patientswithabackgroundhistoryofchronicthyroiditishasa67- to80-foldgreaterriskfactortodevelopPTLthanthosewithoutthis inﬂammatoryprocess[7].

Similartootherlymphomas,subtypesinthyroidlymphomaare classiﬁedaccordingtohistologicalandimmunologicalfeatures.The thyroidglandcontainsnonativelymphoidtissue;intrathyroidal lymphoidtissue candevelop invarious pathologicalconditions, butmostcommonlyoccursinthesettingofautoimmunethyroidi- tis.Thisacquiredlymphoid tissue bearsa closeresemblanceto mucosa-associated lymphoidtissue andcanevolve toanextra- nodal marginal zone B-celllymphoma [8].The developmentof extranodalmarginalzoneB-celllymphomainthethyroidgland isoftencharacterizedbyanindolentcourse,buttransformationto anaggressivelymphomacanalsooccur[8].Incontrast,anyassoci- ationbetweenHodgkin’slymphomaandunderlyingthyroiditishas beendifﬁculttodocumentbecauseofthesmallnumberofcases.

A review of the English literature between 1962 and 2005 revealed16casesofthyroidHodgkin’slymphoma,withafemale preponderanceandgenerallyfavourableoutcome similartoour case[9],patientswithHodgkin’slymphomacommonlypresented witharapidlyenlargingthyroidglandasourcase,or athyroid mass,similartothepresentationofnon-Hodgkin’slymphomaof thethyroid.Themassmaycausesymptomsrelatedtocompression orinﬁltrationofthesurroundingneckorgans.Symptomsreﬂecting airwayoresophagealobstructionoccurredin9/16ofthepreviously reportedcases,butthissignswerenotreportedbythepatient.

On physical examination, the thyroid mass was commonly describedasbeingharduponpalpation(Table1).

Ultrasonographyisusuallythefirstimagingmodalityperformed intheevaluationofa thyroidmass.Previousstudieshavecate- gorizedultrasoundfindingsintothreecategories:diffuse,nodular ormixed.Ithassignificantoverlapwithanaplasticthyroidcan- cer[10–12].Thepresence ofsignificantinternalvascularityand absenceofcalcificationsmaybedistinguishingfeaturesbetween thyroidlymphomaandanaplasticthyroidcanceronultrasonogra- phy[13].

ThediagnosisofHodgkinthyroidlymphomaisoftenpostponed byitsprolongedindolentevolutionwhichdoesnotalwaysappear clear.AlthoughFNAhasbecometheprocedureofchoiceforthe diagnosisofanythyroidtumorithasyieldedmixedresultsassert- ingthepresenceofMALTsothatcoreoropenbiopsyandeven surgicalexcision(asinourcase)isdecisivefordiagnosis[14].

Combinedpathologyandimmunohistochemistrymayspecify microscopy of theselesionsorientingtherapeutic planningand predictingprognosisofthepatients.[15]

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Table1

ClinicopathologicfeaturesofpreviouslyreportedcasesofthyroidHodgkin’slymphoma.

Authors Age(yrs)/sex Presentingsymptoms Imaging/Intraoperative ﬁndings

Pathologicﬁndings Stage Treatment/Outcomes

EvidenceofHL HLsubtypes Uninvolvedthyroid

Ruppetal. 64/M Neckpain,dysphagia

andBsymptoms

Diffuseﬁrmlyenlarged thyroid

Image,description,LN Biopsy

Notreported Lymphocytic Thyroiditis

IIE Unknown

Robertsetal. 61/M Painfulenlargingneck mass

Firmtumormass extendinginto mediastinumand invadingtracheaand carotidsheath

Image,description NS Hashimoto’s

Thyroiditis

IIE Subtotalneckmass

Gibsonetal. 59/F 6yearshistoryof

painlessprogressive neckenlargement

Softtissuswellingwith trachealdeviation

Image,description,LN Biopsy

NS Lymphocytic

Thyroiditis

IIE thyroidectomythen XRT,NEDat6mo.

DeBeatsetal. 57/F 6yearshistoryof goiters,withrapid painfulenlargementof thyroid

Soft-tissueswelling Image,description NS Notdescribed IIE Subtotal

thyroidectomy,no othertherapy,Nedat6 mo.

Feiginetal. 64/F 2yearshistoryofsmall thyroidnoduleand hypothyroidism, hoarsenessandweight loss

Nomediastinal lymphadenopathyor lungdesease

Image,description NS Lymphocytic

thyroiditis

IIE Thyroid

lobectomy,chemoand XRT,NEDat3years.

Kugleretal. 27/F Slowthyroid

enlargementover1yr, stridor,dysphagiaand hoarsenessover3yr.

Thyroidmass contiguouswith mediastinalmass.

Trachealdeviationand narrowing

Image,description NS Notdescribed IIIE Subtotalresectionand

chemo,noFU

Mateetal. 60/F 1yrhistoryof

hypothyroidism,then dysphagiaand hoarseness

Massinvolvingcricoid cartilage,oesophagus andmediastinum.

LNbiopsy MC Notdescribed IIE XRTalone,reccuredin

lungafter6mo,treated withchemo

Mateetal. 25/F Enlarginggoiterand

dysphagia

Thyroidmass compressingtrachea anddisplacing oesophagus

LNbiopsy NS Notdescribed IIE XRTalone,reccured2

yrslaterinabdomen, treatedwithchemo, NEDat7yr.

Smithetal. 19/F 5yrhistoryofgoiter, thendysphagia, dyspneaandrapidly enlargingthyroid

Firmthyroidadherent totracheaand oesophagus

Description NS Notdescribed IIE Unknown

Granadosetal. 36/F 1yrhistoryofgoiter, hypothyroidism.

Increaseinsizeand ﬁrmnessdespite

Bulkymediastinal mass,inﬁltrating thyroidwithtracheal narrowing.

RScellsCD15+ NS Notdescribed IIE XRTalone,NEDat1yr.

Vailatietal. 29/F 2mohistoryofthyroid enlargement,stridor, low-gradefever, fatigueandpruritus.

Homogeneousmass involvingisthmusand leftlobe,withtracheal displacement

Image,description NS Notdescribed IE Subtotal

thyroidectomy,and XRTNEDat2yr.

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M.M.Tatarietal./InternationalJournalofSurgeryCaseReports37(2017)134–138137

Table1(Continued)

Authors Age(yrs)/sex Presentingsymptoms Imaging/Intraoperative ﬁndings

Pathologicﬁndings Stage Treatment/Outcomes

EvidenceofHL HLsubtypes Uninvolvedthyroid

Jayaram 53/F Neckswellingfor2mo,

thencervical lymphadeno-pathy, and

hepatospleno-megalie

6cmﬁrmnoduleinleft lobe,nomediastinalor hilarlymphadenopathy

description,LNbiopsy Notreported Notdescribed IV thyroidlobectomy,lost toFU.

Hardoffetal. 20/F 1yrhistoryofsolitary painlessthyroid nodule,thendeveloped cervical

lymphadenopathy.

cervical,axillaryand mediastinal lymphadenopathy.

RScellsCD15+,CD30+. NS Notdescribed IIE ChemoandXRT,NED

at1yr.

Hardoffetal. 18/F Painlessthroatfullness, dysphagia,fever.

Solitaryleftthyroid nodule,mediastinal andcervical lymphadenopathy.

RScellsCD15+,CD30+. NS Notdescribed IIE ChemoandXRT,NED

at18mo.

Luboshitzkyetal. 19/F Singlenoduleinleft lobeofthyroid.

5cmmediastinalmass andanenlarged cervicallymphnode

RScellsCD15+,CD30+. NS Notdescribed IIE NEDat2yr.

Nakamuraetal. 18/M Progressivelyenlarging neckmassanddyspnea

Largemassinvolving entirethyroid extendinginto mediatinum

RScellsCD15+,CD30+. NS Notdescribed IIE XRTalone,NEDat4yr.

Tatarietal. 65/M 8morapidlycervical

massincreasing, painless,general pruritis,nightsweats, fatigue

tissuemassoftheright thyroidlobe,dippingto theanterioretmiddle mediastinum

Image,description NS Notdescribed IIE Totalethyroidectomie

andchemo.

Yr=year;mo=month;wk=week;HL=Hodgkin’slymphoma;LN=lymphnode;RS=Reed–Sternberg;NS=nodularsclerosissubtype;MC=mixedcellularitysubtype;XRT=radiationtherapy;NED=noevidenceofdisease;

FU=follow-up;chemo=chemotherapy.

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Hodgkin’s lymphoma is characterized by the presence of Reed–Sternbergcells.Thisisconsistentwiththewell-recognized tendencyforscleronodularsubtypetooccurinthemediastinum and head and neck region [16]. In many cases, the associated ﬁbrosis and sclerosis were more pronounced within the thy- roidglandincomparisontotheadjacentnodalareas,mimicking theﬁbrosclerosingvariantofHashimoto’sthyroiditisorinsome casesReidel’sthyroiditis,andrequiringcarefulhistologicexami- nationandimmunohistochemicalanalysistomakethediagnosis ofHodgkin’slymphoma.

Mostof thethyroidHodgkin’slymphomapatientspresented withlow-stagediseaserespondedtochemotherapywithorwith- out radiation therapy and had a favorable clinical outcome.

Surgicalinterventionisrarelyrequiredinthetreatmentofnodal Hodgkin’slymphoma.However,inthyroidHodgkin’slymphoma, somepatientspresentedwithsymptomsthatcompromisedtheir airwayorcausedseverepain,requiringsurgicaltherapy topal- liatetheirsymptoms.Surgicalinterventiondidnotappeartobe associatedwithadverseoutcomeinthyroidHodgkin’slymphoma patients,similartothyroidnon-Hodgkin’slymphomapatients[17].

Ourcasehighlightsthevalueoftotalthyroidectomyinthyroid Hodgkin’slymphomaforclinicalandhaematologicalresolution.

4. Conclusion

Since1962,onlysixteencasesappeartohavebeendescribedin theliterature,thatshowtheinterestofthepresentedcase.

Theparticularityofthiscaseisthefactthatitisaboutaman,and theisolatedandprimarylocalisationinthethyroidgland.Allsymp- tomshavedisappearedaftertotalthyroidectomyandfewcuresof chemotherapy.Noreccurencewasnotedafteratwoyearsfollow -up.

Conﬂictsofinterest

Theauthorsdeclarehavingnoconﬂictsofinterestforthisarticle.

Sourcesoffunding None.

Ethicalapproval

Writteninformedconsentforpublicationoftheirclinicaldetails and/orclinicalimageswasobtainedfromthepatient.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimage.

Authorcontribution

MohammedMoutaaTatari:Correspondingauthorwritingthe paper

SaidAnajar:writingthepaper RedaAbada:studyconcept SamiRouadi:studyconcept

MohammedRoubal:correctionofthepaper MohammedMahtar:correctionofthepaper

Registrationofresearchstudies researchregistry2462.

Guarantor

DRTatariMohammedMoutaa.

Acknowledgement None.

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