• 沒有找到結果。

Multiple calcifications within the parotid gland of patients with Sjögren’ssyndrome Oral Science International

N/A
N/A
Protected

Academic year: 2022

Share "Multiple calcifications within the parotid gland of patients with Sjögren’ssyndrome Oral Science International"

Copied!
5
0
0

加載中.... (立即查看全文)

全文

(1)

Multiple calcifications within the parotid gland of patients with Sjögren’s syndrome

Masahiro Izumi

, Yoshiaki Kise, Keiko Murata, Atsushi Murata, Miwa Nakayama, Yoshiko Ariji, Munetaka Naitoh, Eiichiro Ariji

DepartmentofOralandMaxillofacialRadiology,Aichi-GakuinUniversitySchoolofDentistry,Nagoya,Japan

a r t i c l e i n f o

Articlehistory:

Received21March2012

Receivedinrevisedform24May2012 Accepted1June2012

Keywords:

Sjögren’ssyndrome Calcinosis Parotidgland

Multidetectorcomputedtomography

a b s t r a c t

Purpose:Thepurposeofthisstudywastoinvestigatecomputedtomography(CT)andclinicalfeatures relatingtocalcificationswithintheparotidglandofpatientswithSjögren’ssyndrome(SS).

Methods:Datafrom30patientswithSSwhohadbeenexaminedbyCTwereextractedfromourradiolog- icalinformationdatabaseaccumulatedfrom2001to2011,andtheirCTimageswererereadcarefully.Of thesepatients,14(allfemale;agerange20–95years;meanage61.4years)withcalcificationswithinthe parotidglandwereretrospectivelyinvestigatedwithCTfindings.Therelationshipbetweencalcification occurrenceandclinicalsymptomsincludingparotidswellingand/orsalivacolicwasinvestigated.The degreeofdestructionoftheparotidglandonCTimageswasalsoevaluated.

Results:Allcalcificationsof14patientswerelocatedwithintheparotidgland,notintheparotidduct.CT imagesofallcalcificationsshowedsmallandregularroundshapes.Multipleoccurrencesofcalcifications wererecognizedin10patients,andasolitaryoccurrencewasseenin4patients.Sevenpatientshad bilateralcalcifications.Therewaslittlerelationshipbetweentheoccurrenceofcalcificationsandclinical symptoms,andtheseverityofdestructionoftheparotidgland.

Conclusion:ThepresentedCTandclinicalfeatureswouldbepeculiartoSSbecausetoomanypatients lackedthetypicalfeaturesofsialolithswithintheparotidgland.

© 2012 Japanese Stomatological Society. Published by Elsevier Ltd. All rights reserved.

1. Introduction

Sjögren’ssyndrome(SS)isamulti-systemautoimmunedisor- dermainlytargetingthesalivaryandlacrimalglands[1–4].Clinical symptomsarecharacterizedbyprogressivedrymouthanddryeyes [2–4].Inrecentyears,bilateralandmultiplesmallcalcificationsin theparotidparenchymahavebeenreportedasanewfeatureofSS [5–8].Theoccurrenceofsmallcalcificationswasconsideredtobe intheseverelydestructedparotidparenchymaofSS[8]andtobe extremelyrareinthewholeSSpopulation[6,7].Toourknowledge, therehadbeenlessthan20casesinthepreviousnumerousstudies ofSS[5–8].Therefore,characteristicsofsmallcalcificationswere notsufficientlyinvestigated.

Severalimagingmodalities, suchasplainX-rayexamination [8,9], sialography, ultrasound, and computed tomography (CT) [5,10],areavailablefordetectionofcalcifications intheparotid gland.CTisthemostusefultoolindetectionandevaluationofsmall calcificationsbecauseofitshighspatialresolution[11,12].CTcan

∗ Correspondingauthorat:DepartmentofOralandMaxillofacialRadiology,Aichi- GakuinUniversitySchoolofDentistry,2-11Suemori-Dori,Chikusa-Ku,Nagoya464- 8651,Japan.Tel.:+81527592165;fax:+81527592165.

E-mailaddress:izumim@dpc.agu.ac.jp(M.Izumi).

alsodepictthedestructedparotidparenchyma,whichwasreplaced byfat,duetoautoimmunereactionsofSS[8,13].Weplannedaret- rospectiveCTinvestigationtoclarifysomecharacteristicsofsmall calcifications.ThepurposeofthisstudywastoinvestigateCTand clinicalfeaturesrelatingtocalcificationswithintheparotidgland ofpatientswithSS.

2. Patientsandmethods

WesurveyedpatientswithSS,whohadbeenexaminedbyCT, fromaradiologicalinformationdatabaseaccumulatedfrom2001to 2011.Datafrom38patientswithSSwereextracted.Thepurposesof CTexaminationwerefordiagnosisofinflammation,cysts,tumors, andtrigeminalneuralgia.PatientshadconsentedtoCTexamina- tionanditsstudyapplication.CTimageswererereadcarefullyto confirmthepresenceofcalcificationsintheparotidglandbythree radiologists.Clinicalinformationwassimultaneouslyinvestigated frommedicalrecordsandlettersofintroductionfrompreviousdoc- tors,inparticularpastexaminationsfordefinitediagnosisofSS, complicationsofotherautoimmunediseases,ahistoryofparotid swelling,and/orsalivacolic.Ifclinicalinformationwasnotenough, we interviewed the patientsagain if possible. Allpatients sat- isfiedtherevisedJapanese diagnosticcriteriaforSSof 1999.Of 1348-8643/$seefrontmatter © 2012 Japanese Stomatological Society. Published by Elsevier Ltd. All rights reserved.

http://dx.doi.org/10.1016/S1348-8643(12)00052-3

(2)

Fig.1.TheseverityofdestructionoftheparotidglandparenchymainpatientswithSjögren’ssyndrome.(A)Slightdestruction;Slightfatdepositsareseenwithintheparotid gland,andmostparenchymaremain.(B)Moderatedestruction;Moderatefatdepositsareseenwithintheparotidgland,andparenchymaremaintoadegreebetweenslight andsevere.(C)Severedestruction;Severefatdepositsareseenwithintheparotidgland,andmostparenchymadisappear.

38patients,8patientswereexcludedbecause5hadsialographic examinationbeforeCTexaminationand3patientshadinsufficient clinicalinformationforthisstudy.Oftheremaining30patients, 14(allfemale;agerange20–95years;meanage61.4years)had calcificationsintheparotidglandonCTimages.

CT examination was performed with a single slice scanner (Somatom ART:Siemens Medical Systems, Erlangen,Germany), a 2-detectorscanner (HiSpeedNX/I Pro:GEYokogawa Medical Systems,Tokyo,Japan),ora4-detectorscanner(Asteion:Toshiba MedicalSystemsCorporation,Tokyo,Japan).Scanconditionswere 120kVand100mA.Transverseimageswith3mmor2mmthick- nesswerecontinuouslyacquiredwithascandirectionparallelto theocclusalormandibularplane.

CTimagesof14patientswithcalcificationswereinvestigated forlocation,distribution,size,andshapeofcalcifications.Thesever- ityofthedestructedparotidparenchymaonCTimageswasalso evaluatedbasedonaconsensusoftwoexperiencedradiologists.

Wepresumedthedestructedparotidparenchyma,whichresults fromautoimmunereactionsofSS,fromtheextentoffatdeposition

intheparotidgland(Fig.1).WhethertheseCTfindingsandclinical featureshadanycharacteristicsin14patientswereinvestigated.

3. Results

CTfindingsandclinicalfeaturesofthe14patientsaresumma- rizedinTable1.Thereweremanycharacteristicsdifferingfromthe typicalfeaturesofsialoliths.

Allcalcifications of the 14 patientswerelocated withinthe parotidgland,notintheparotidduct.CTimagesofallcalcifications showedthatthesizedidnotexceed2mmandtheshapewasregu- larandround.Multipleoccurrencesofcalcificationswereobserved in10patients,andbilateraloccurrencewasseenin7patients.

As to the relationship between the occurrence of calcifica- tionsandtheseverityofthedestructedparotidparenchyma,slight destructionamountedtoabout60%.Therewereafewpatientswith ahistoryofparotidswellingand/orsalivacolic(21%,3/14)andwith complicationsofotherautoimmunediseases(14%,2/14).

(3)

4 80 WithinBil.PG Multiple <2mm Regularround Severe RA

5 58 WithinBil.PG Multiple <2mm Regularround Slight

6 20 WithinLt.PG Multiple <2mm Regularround Slight

7 42 WithinRt.PG Multiple <2mm Regularround Slight

8 68 WithinBil.PG Multiple <2mm Regularround Slight

9 71 WithinBil.PG Multiple <2mm Regularround Slight +

10 41 WithinBil.PG Multiple <2mm Regularround Slight +

11 95 WithinLt.PG Solitary <2mm Regularround Moderate RA

12 40 WithinRt.PG Solitary <2mm Regularround Moderate

13 55 WithinRt.PG Solitary <2mm Regularround Slight

14 64 WithinRt.PG Solitary <2mm Regularround Severe +

Rt,right;Lt,left;Bil,bilateral;PG,parotidgland;–,None;RA,rheumatoidarthritis.

Fig.2showstypicalmultiplecalcificationswithinthebilateral parotidgland.Manysmallcalcificationswerescatteredthroughout thewholeoftheparotidgland.Fig.3shows anatypicalcaseof calcificationswhereisolatedcalcificationwasobservedwithinthe rightparotidgland.

4. Discussion

SSisamulti-systemautoimmunedisorderthatcausesabnor- malitiesinmanyorgansandtissues.Theoccurrenceofcalcifications inSShasbeencomparativelyknownasoneoftheextra-glandular

Fig.2. Computedtomography(CT)imagesofa55-year-oldwomanwithSjögren’ssyndrome(CaseNo.3).(A)AxialCTimageforsofttissuewindowshowsatypicalfinding ofbilateralmultiplecalcificationsintheparotidgland.(B)AxialCTimageforbonewindowofthesameslice.(C)ThreedimensionallyreconstructedCTimageclearlyshows scatteredcalcificationsintherightparotidgland.

(4)

Fig.3. Computedtomography(CT)imagesofa40-year-oldwomanwithSjögren’s syndrome(CaseNo.12).AxialCTimageforsofttissuewindowshowsanatypical findingofsolitarycalcificationintherightparotidgland(arrow).

manifestations,suchasnephrolithiasisand urolithiasis[14–16].

Distalrenaltubularacidosisisconsideredasariskfactorfordevel- opmentofcalcifications[14–16].

Incontrast,thecalcificationswithinthebilateralparotidgland ofSS werenotwellknownbecauseof theirrareoccurrence in SS[6,7].However,consideringtheincidenceofourinvestigation, calcificationswithinthebilateralparotidglandappeartobeafre- quent occurrence (23%,7/30). Sun etal. also reportedthat the incidenceofcalcificationswithinthebilateralparotidglandofSS was29.4–35.2%intheirCTinvestigation[8].Areasonforthecon- tradictionbetweenrareandfrequentoccurrencemaybethatCT examinationisseldomdoneinpatientswithSS.Mostradiological examinationstoSSarescintigraphyand/orsialography,whichare inferiortodetectsmallcalcifications,fordefinitediagnosisofSS.

IfCTexaminationisperformedinhigherfrequency,thepotential existenceofcalcificationswithintheparotidglandmayberevealed inmanypatientswithSS.

Mostpreviousreportsdealtwiththesecalcificationsassialoliths [5–7]. However, in this investigation,a lack of CT and clinical features possibly relating to sialolith occurrence wasobserved.

Ordinarysialolithsoccuratanincidencerateofabout1%inthe population[17],and 6–20%of thewholesialolithsoccurinthe parotidgland[10].InourSSseries,theincidencerateofcalcifi- cationswithintheparotidglandwas46%(14/30).Furthermore, althoughsialolithsarepredominantly foundintheduct,allcal- cificationsinourcaseswerefoundwithinthegland.Theshape of ordinary sialolithsis oblong and is often pointed and sharp [17]. Thesecalcifications were regularand roundshapes. Com- monly,sialolithiasispresentswithpainfulswelling(59%),painless swelling(29%),andpainonly(12%)[10].Clinicalsymptomsinour calcificationcaseswerefew(21%,3/14).Althoughlowersalivary flow rate is considered to be associated with sialolith occur- rence [10], the severity of the destructed parotid parenchyma was not related to the occurrence of calcifications in our SSseries.

RauchandGorlindescribedinthetextbook[17]oforalpathol- ogythat“The“calculus”ofthesalivaryglandisetiologicallyand clinicallyasarelativelyheterogeneousterm.Apartfromsialolith, therearedysplasticcalcificationsininflammatorysalivarygland tissues and calcified venousthrombi.” Asthey described, some calcificationsinthisinvestigationmaybedysplasticcalcifications ofthesalivaryglandtissuessecondarytochronicinflammation of SS. And CT findings of these calcifications were very simi- lartothecharacteristicsofangioliths[18]in theparotidgland.

Regardingthecalculusofthesalivarygland,we shouldkeep in mind the existence of calcifications except for sialolith in the duct.

Wedonot negateanotionofpreviousreportsthat allcalci- ficationsinSScasesaresialoliths,becauseofnoclearorenough evidencetonegatethisnotionatpresent.Andthisinvestigation hasthefollowinglimitations:asmallnumberofcases,casebias, andnon-analysisofthecomponentsofcalcifications.Somecontra- dictionsbetweenordinarysialolithsandthesecalcificationsmaybe astudyerror.Butitisdifficulttoconsiderfromouroverallresults thatthesecalcificationsoccurredfromjustthesamemechanismas ordinalsialolithsintheducts.Furtherresearchbypathophysiolog- icalandbiochemicalapproachesisexpectedtorevealthisissuein thefuture.

Fig.4. Computedtomography(CT)imagesofa57-year-oldmanwithSjögren’ssyndrome.Hehad7yearshistoryofsialographicexaminationoftherightparotidglandbefore CTexamination.(A)High-densitystructuresintheductsareconsideredtobetheresidueofcontrastmedium(arrow).(B)Ontheinferiorslice,high-densityspotscannotbe distinguishedfromcalcifications(arrow).

(5)

differ fromordinary sialoliths in the mechanism of occurrence because too many cases lacked typical features of ordinary sialoliths.

Conflictofintereststatement

Noneoftheauthorshasanyconflictofinterestregardingthis research.

References

[1]Delaleu N, Jonsson R, Koller MM. Sjögren’s syndrome. Eur J Oral Sci 2005;113:101–13.

[2]Gannot G, Lancaster HE, Fox PC. Clinical course of primary Sjögren’s syndrome: salivary, oral, and serologic aspects. J Rheumatol 2000;27:

1905–9.

[3] KruizeAA,HenéRJ,vanderHeideA,etal.Long-termfollowupofpatientswith Sjögren’ssyndrome.ArthritisRheum1996;39:297–303.

[4]PijpeJ,KalkWW,BootsmaH,etal.Progressionofsalivaryglanddysfunctionin patientswithSjögren’ssyndrome.AnnRheumDis2007;66:107–12.

[9] SuleimanSI,HobsleyM.Radiologicalappearancesofparotidductcalculi.BrJ Surg1980;67:879–80.

[10] WilliamsMF.Sialolithiasis.OtolaryngolClinNorthAm1999;32:819–34.

[11]BryanRN,MillerRH,FerreyroRI,etal.Computedtomographyofthemajor salivaryglands.AmJRoentgenol1982;139:547–54.

[12]MandelL,HatzisG.Theroleofcomputerizedtomographyinthediagnosisand therapyofparotidstones:acasereport.JAmDentAssoc2000;131:479–82.

[13] IzumiM,EguchiK,NakamuraH,etal.Prematurefatdepositioninsalivary glandsaffectedbySjögren’ssyndrome:evidencebyMRandCT.AmJNeuroradiol 1997;18:951–8.

[14]Moutsopoulos HM,Cledes J, SkopouliFN,et al. Nephrocalcinosis inSjö- gren’s syndrome: a late sequela of renal tubular acidosis.J Intern Med 1991;230:187–91.

[15]ErikssonP,DennebergT,TiseliusHG.Riskfactorsofcalciumstoneformation inpatientswithprimarySjögren’ssyndrome.UrolRes1996;24:39–43.

[16]ErikssonP,DennebergT,LarssonL,etal.Biochemicalmarkersofrenaldisease inprimarySjögren’ssyndrome.ScandJUrolNephrol1995;29:383–92.

[17]RauchS,GorlinRJ.Diseasesofthesalivaryglands.In:GorlinRJ,GoldmanHM, editors.Thoma’soralpathologysixthedition,vol.2.Missouri:C.V.MosbyCo;

1970.p.996–1003.

[18]RaymondAK,BatsakisJG.Angiolithiasisandsialolithiasisintheheadandneck.

AnnOtolRhinolLaryngol1992;101:455–7.

[19] SchortinghuisJ,PijpeJ,SpijkervetFK,etal.Retentionoflipiodolafterparotid glandsialography.IntJOralMaxillofacSurg2009;38:346–9.

參考文獻

相關文件

pylori in pathogenesis of oral mucosal lesions or ulcerations is still unclear, it seems that patients with oral lesions as leukoplakia and oral lichen planus, and concurrent

The length of SP when exceeds 30 mm it is said to “elongated.” The purpose of this retrospective study was to assess the prevalence of elongated SP (ESP) by cone beam

E ffective utilization of dental and zygomatic implants may help to restore oral function in patients with severe maxillary defects.. This clinical report describes the management

The aim of this study was to investigate, through a prospective clinical study, the prevalence and characteristics of oral lichen planus (OLP) and lichenoid lesions (OLL) in

Therefore, the objective of this work is to diagnose incipience lesions of OHL by using in situ hybridization for identification of EBV within the oral mucosa of HIV- infected

In this respect, the aim of the present study was to as- sess volumetric as well as morphological surface changes of the orbital cavity in patients treated with both tooth- borne

This case was challenging because the largely undifferen- tiated morphological appearance was difficult to distinguish from a high-grade salivary neoplasm (e.g. neuroendocrine

Salivary gland adaman- tinoma-like Ewing sarcoma is rare; the majority of reported cases involve the parotid gland, followed by the submandibu- lar gland, and occur in older