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Pleomo phic adenoma o he sublingual gland: A case epo
D iss Elid issi 1, *, Yousse Lakhda 1, Mohamed Ya i 1, Oma Oulghoul 1, Mohamed Chehbouni 1, O hman
Benhoummad 2, Yousse Rochdi 1 and Abdelaziz Raji 1
1 Depa men o ENT and HNS Su ge y, Mohammed VI Uni e si y Hospi al, Ma akech, Mo occo.
2 Depa men o ENT and HNS Su ge y, Facul y o Medicine and Pha macy o Agadi , Ibn Zoh Uni e si y, Agadi , Mo occo.
Wo ld Jou nal o Ad anced Resea ch and Re iews, 2025, 26(02), 1462-1466
Publica ion his o y: Recei ed on 28 Ma ch 2025; e ised on 09 May 2025; accep ed on 11 May 2025
A icle DOI: h ps://doi.o g/10.30574/wja .2025.26.2.1712
Abs ac
Pleomo phic adenoma, also known as mixed umo , is he mos common neoplasm a ising om sali a y glands,
accoun ing o app oxima ely 81.2% o all benign umo s in sali a y glands. The pa o id gland accoun s o 85% o hese
umo s, wi h 10% a ise in he submandibula gland and only 0.3% in he sublingual gland. The aim o his s udy is o
desc ibe he speci ic managemen o pleomo phic adenoma in i s unusual loca ion. He e, we p esen a case o
pleomo phic adenoma o he sublingual gland in a 62-yea -old woman, mani es ing wi h a painless mass in he loo o
he mou h o 5 yea s, appea ing as a well-limi ed hypodense issue lesion, enhanced a e con as injec ion, in he
sublingual egion and ex ending o he igh mylohyoid muscle in compu ed omog aphy imaging. The umo and
sublingual gland we e esec ed unde gene al anes hesia. The his opa hological diagnosis was pleomo phic adenoma.
No e idence o ecu ence was de ec ed a e h ee yea s ollow-up.
Keywo ds: Pleomo phic Adenoma; Sali a y Gland Neoplasm; Sublingual Gland
1. In oduc ion
Sali a y gland umo s a e a e and accoun o 3.5-10% o all head and neck umo s. Tumo s o he sublingual sali a y
glands accoun ing o 0.3-5.2% o all epi helial sali a y gland umo s and app oxima ely 1.5% o all ca cinomas o he
main sali a y glands [1, 2]. Mos umo s a ising om he sublingual gland a e malignan , wi h an es ima ed p e alence
o 80-90% [3, 4]. In con as , benign umo s such as pleomo phic adenoma (PA) a e a ely epo ed [5-6]. Due o he
a i y o PA in he sublingual gland, mos obse a ions om a ious epo s lack de ailed compa isons o pa hological
indings and long- e m ou comes. He e, we p esen ou own case and p o ide a comp ehensi e e iew and discussion
o he exis ing li e a u e.
2. Case epo
A 62-yea -old woman wi h no medical his o y p esen ed wi h a painless mass in he igh loo o he mou h ha had
been p esen o 5 yea s. Examina ion o he o al ca i y e ealed a well-bounded, mobile, i m, lobula ed mass o he
igh buccal loo co e ed wi h no mal o al mucosa, measu ing 4 cm x 3 cm. The mass was no a ached o su ounding
s uc u es, including he mandible (Figu e 1). Sali a low om he igh submandibula canal was undis u bed. The
pa ien had poo den al condi ion wi h no signs o local in ec ion, ongue mobili y and as e we e no mal. The e was no
acial asymme y o ce ical adenopa hy, and he es o he ENT examina ion was un ema kable.
The compu ed omog aphy scan e ealed a ela i ely well-de ined, hypodense umo , enhanced ollowing con as
injec ion, in he sublingual egion and ex ending o he igh mylohyoid muscle. The umo was measu ed a
43.5x45x26.5mm (Figu e 2).
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The umo was excised using an in a-o al app oach unde gene al anes hesia. A longi udinal incision was made in he
mucosa o he loo o he mou h opposi e he umo , which was hen dissec ed wi h he sublingual gland in o a single
mass minu ely om he muscula and ascula s uc u e o he loo o he mou h (Figu e 3). The umo did no adhe e
o he su ounding issue.
The su gical p ocedu e was success ul in main aining hemos asis, and no in aope a i e o pos ope a i e complica ions
we e epo ed. These included he absence o hema oma o he loo o he mou h and no pa alysis o he ongue o as e
p oblem.
His ological examina ion e ealed he p esence o a sali a y pa enchyma con aining an encapsula ed umo
p oli e a ion, which consis ed o wo dis inc con ingen s: an epi helial and a s omal componen . The p oli e a ion is
o ganized in o lobules, which a e sepa a ed by a hyaline and disc ee ly myxoid s oma. This is compa ible wi h a
diagnosis o pleomo phic adenoma (Figu e 4).
The clinical and CT scan ollow-up was un ema kable, wi h no e idence o umo ecu ence a e h ee yea s ( igu e
5).
Figu e 1 Righ buccal loo mass co e ed by in ac o al mucosa
Figu e 2 CT scan wi h Axial (A) and Sagi al (B) cu s showing a ai ly well-limi ed, hypodense umo o he sublingual
gland ex ending o he igh mylohyoid muscle
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Figu e 3 Su gical excision piece
Figu e 4 Tumo wi h a dual epi helial and mesenchymal composi ion, wi h a myxoid s oma (magni ica ion X 100,
Hema ein Eosin s ain)
Figu e 5 Pho o o he buccal loo one yea a e su ge y showing he absence o umo ecu ence
3. Discussion
Sali a y gland umo s accoun o only 3.5-10% o head and neck umo s, and umo s o he sublingual glands a e he
leas common compa ed wi h umo s o he pa o id, submandibula and mino sali a y glands [7]. The majo i y o hese
umo s a e malignan , and a ew benign cases o pleomo phic adenomas ha e been desc ibed in he sublingual glands
[6-8]. The e o e, when a umo o he sublingual glands is suspec ed, as in he p esen case, i is essen ial o ake in o
accoun he high p obabili y o malignancy.
PA is he mos common ype o sali a y gland umo , wi h a p e alence o 81.2%. The si es mos suscep ible o
umo igenesis a e he pa o id glands (85%), ollowed by he submandibula glands (6-11%), he mino sali a y glands
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(10-23%) and he sublingual glands (0.3-2%) [1-9]. PA is ypically sligh ly mo e common in women, and age o onse is
mo e widely dis ibu ed han in o he sali a y gland umo s [5]. Clinically, an asymp oma ic swelling in he loo o he
mou h is he mos common complain . Tumo s o he sublingual sali a y gland a e gene ally only ecognized a an
ad anced s age, mainly because o hei minimal symp oma ology. In e ms o disease du a ion, umo g ow h ends o
be slow, and subjec i e symp oms such as ulce s, pain, and unc ional impai men a e a e, so i ends o go un ea ed
o ela i ely long pe iods.
Imaging is essen ial in he p eope a i e assessmen o PA. On CT and MR images, PA p esen as well-de ined lesions
occasionally accompanied by cha ac e is ic lobula ed con ou s. On T2 weigh ed images, ypical PA show ma ked
hype in ensi y, which e lec s he abundan myxochond oid s oma, wi h a hypoin ense im indica ing he ib ous
capsule. Howe e , in a umo ally signal in ensi y a ies acco ding o he cellula densi y, p opo ion o epi helial and
s omal componen s, and ype o s omal componen s [10].
Incision biopsy o la ge sali a y gland umo s is o en c i icized due o he isks o umo cell dissemina ion and ne e
damage and he p ocedu al di icul ies posed by umo adhesion [11,12]. Fo hese easons, ine-needle aspi a ion
biopsy (FNAB) is equen ly pe o med because i is a simple and minimally in asi e p ocedu e [13]. Howe e , FNAB
has low diagnos ic accu acy o sali a y gland umo s due o he a ie y o his ological ea u es and mul i ude o
his ological ypes, and samples a e poo due o he low cell sampling coun [13,15].
Ea ly o al su gical esec ion o he sublingual gland and i s neoplas ic mass in no mal ma gins o benign umo s is he
ea men o choice o a oid ecu ences. The excision may possibly be accompanied by selec i e neck dissec ion and
adio he apy o malignancies. The his ologic examina ion o he lesions is necessa y no only o es ablish he diagnosis
bu also o he be e managemen o he su ounding issues [15]. Depending on he umo size and loca ion, he o al
esec ion may become qui e complex due o he close p oximi y o he sublingual gland o he inne co ex o he
mandible, he submandibula sali a y gland and i s duc , he lingual essels and ne e, and he hypoglossal ne e [8,
15]. In ou case, clinical and CT imaging ea u es indica ed a close- o-su ace, ci cumsc ibed, easily accessible umo ,
and hus an in ao al o al esec ion o he lesion was pe o med.
Pleomo phic adenomas a e gene ally benign umo s. I can ecu as well as i can gi e ise o a malignan
ans o ma ion. Long- e m clinical and adiological moni o ing, las ing o yea s, is essen ial a e o wa ch ou o hese
complica ions.
4. Conclusion
The pleomo phic adenoma is a he e ogeneous benign umo combining a ious epi helial and mesenchymal s uc u es
epi helial and mesenchymal s uc u es. I is less common in he accesso y sali a y glands, pa icula ly he sublingual
gland. Because o he isk o local ecu ence and malignan degene a ion, all pa ien s unde going su ge y o a
pleomo phic adenoma mus unde go egula clinical moni o ing.
Compliance wi h e hical s anda ds
Disclosu e o con lic o in e es
The e a e no con lic s o in e es o decla e ela ed o his esea ch
S a emen o e hical app o al
While o mal e hical app o al was no ob ained o his s udy, we ensu ed ha all aspec s o he esea ch we e
conduc ed e hically and wi h espec o he igh s and well-being o he pa icipan s.
S a emen o in o med consen
In o med consen was ob ained om all pa icipan s in ol ed in he s udy, and his in o ma ion has been app op ia ely
included in he manusc ip .
Funding
This esea ch ecei ed no ex e nal unding.
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