J Clin Exp Den . 2017;9(1):e167-71. Ke a ocys ic odon ogenic umo
e167
Jou nal sec ion: O al Medicine and Pa hology
Publica ion Types: Case Repo
Maxilla y pe iphe al ke a ocys ic odon ogenic umo . A clinical case epo
Ma ía del Ca men Vázquez-Rome o 1, Ma ía de los Angeles Se e a-Figallo 1, Ja ie Albe di-Na a o 2, Ja ie
Cabezas-Tala e o 3, Manuel-Ma ía Rome o-Ruiz 1, Daniel To es-Laga es 1, Jose-Manuel Agui e-U iza 2,
Jose-Luis Gu ié ez-Pé ez 1
1 Mas e ’s Deg ee in O al Su ge y - School o Den is y - Uni e si y o Se ille
2 Mas e ’s Deg ee in O al Pa hology - Depa men o S oma ology II UFI11/25 School o Medicine and Den is y - Uni e si y o
he Basque Coun y/EHU
3 P i a e p ac ice - Cáce es, Spain
Co espondence:
O al Su ge y Depa men
Facul y o Den is y. Se ille, Spain
C/ A icena s/n. 41009. Se ille, Spain
[email p o ec ed]
Recei ed: 19/08/2016
Accep ed: 31/08/2016
Abs ac
The ke a ocys ic odon ogenic umo is a benign odon ogenic cys ic neoplasia cha ac e ized by i s hin, squamous
epi helium wi h supe icial pa ake a osis. I has he po en ial o in il a ion and local agg essi eness and has a high
a e o ecu ence.
This neoplasia is p edominan ly ound in males and people o whi e o igin. The mandible is he mos equen ly
in ol ed si e, in pa icula he hi d mola egion, mandibula angle, and amus. I has a mandible-maxilla a io o
2:1. Only abou wen y cases o pe iphe al ke a ocys ic odon ogenic umo s (PKCOT) ha e been epo ed in he
in e na ional li e a u e.
This s udy p esen s a case o PKCOT localized in he an e io egion o he maxilla, on he es ibula side o he
uppe le la e al inciso and he uppe le canine. The diagnosis and ea men p ocedu es, as based on he li e a-
u e, a e also discussed.
Key wo ds: Odon ogenic cys s, odon ogenic umo s, ke a ocys , ke a ocys ic odon ogenic umo .
doi:10.4317/jced.53438
h p://dx.doi.o g/10.4317/jced.53438
In oduc ion
The ke a ocys ic odon ogenic umo (KCOT) is a benign
odon ogenic cys ic neoplasia cha ac e ized by i s hin,
squamous epi helium wi h supe icial pa ake a osis. I
has he po en ial o in il a ion and local agg essi eness
and has a high a e o ecu ence (1-3).
This neoplasia is p edominan ly ound in males and
people o whi e o igin. I occu s mainly in he mandible,
in pa icula he hi d mola egion, mandibula angle,
and amus, wi h a mandible-maxilla a io o 2:1.1. I can
appea a any age; howe e , i is mo e equen be ween
he ages o 20 and 30. I s incidence a e anges om 3
A icle Numbe : 53438 h p://www.medicinao al.com/odo/indice.h m
© Medicina O al S. L. C.I.F. B 96689336 - eISSN: 1989-5488
eMail: [email p o ec ed]
Indexed in:
Pubmed
Pubmed Cen al® (PMC)
Scopus
DOI® Sys em
Vázquez-Rome o MC, Se e a-Figallo MA, Albe di-Na a o J, Cabezas-
Tala e o J, Rome o-Ruiz MM, To es-Laga es D, Agui e-U iza JM,
Gu ié ez-Pé ez JL. Maxilla y pe iphe al ke a ocys ic odon ogenic umo .
A clinical case epo . J Clin Exp Den . 2017;9(1):e167-71.
h p://www.medicinao al.com/odo/ olumenes/ 9i1/jced 9i1p167.pd
J Clin Exp Den . 2017;9(1):e167-71. Ke a ocys ic odon ogenic umo
e168
o 12% o odon ogenic umo s (4). Simila ly, his lesion
can appea suddenly as a single clinical en i y o as a
complica ion o Go lin-Gol z Synd ome (3).
Philipsen coined he e m “odon ogenic ke a ocys ic”
(OKC) o he i s ime in 1956 (4). The his opa hologic
c i e ia o diagnosis o OKC we e i s es ablished by
Pindbo g e al. (5) in 1962, in which pa icula a en ion
was paid o i s pa ake a inaza ion. In 2005, he Wo ld
Heal h O ganiza ion (WHO) eclassi ied he OKC as
KCOT because o i s clinical beha io and some gene ic
aspec s (including local agg ession, in il a i e g ow h,
and a high a e o ecu ence o up o 62.5%) (6).
Said umo gene ally occu s in aosseously (1,7) and i
is much less likely o g ow ex aosseously, in less han
0.5% o he cases desc ibed (6). Dayan e al. (8) desc i-
bed he e m pe iphe al odon ogenic ke a ocys (POKC)
in 1988, and i was la e enamed as pe iphe al ke a o-
cys ic odon ogenic umo (PKCOT) (4). Acco ding o
he li e a u e, o da e only 22 cases o PKCOT ha e been
epo ed, wi h hese being p ima ily obse ed on gum
issue (17 ou o 22 cases (8-18)), al hough i can also
occu in he o al mucosa (only h ee cases ha e been
desc ibed in he li e a u e (19,20)) and in he la e al a-
cial deep egion ( wo cases epo ed (21)), wi h an occu-
ence a e o 77%, 14%, and 9%, espec i ely. In some
cases, he lesion is cha ac e ized by an ex aosseous side
and a sligh bone issue in asion, a pe o a ion o he
co ical bone ound unde he PKCOT in some cases.
O he cases a e s ic ly ex aosseous, wi h no mal o al
mucosa co e ing he PKCOT.
This s udy p esen s a case o PKCOT localized in he
an e io egion o he maxilla on he uppe le la e al
inciso and he uppe le canine. The diagnosis and
ea men p ocedu es, as well as he main clinicopa ho-
logical aspec s, a e also discussed.
Case Repo
A 32-yea -old male p esen ed a lump loca ed in he an e-
io egion o he le uppe jaw. The pa ien had no iced
Fig. 1. A) In ao al iew o he lesion B) CBCT: axial sec ion and C) CBCT: sagi al sec ion: lesion loca ion, in-
aosseous and ex aosseous in ol emen .
a clea g ow h and sel - epo ed h ee mon hs o e o-
lu ion. The pa ien ’s p ima y conce n was no only he
inc easing gum size i sel , bu also i s es he ic ami ica-
ions, as he g ow h became isible upon smiling.
The in ao al examina ion e ealed a whi ish lump wi h
a so su ace loca ed be ween he uppe le la e al inci-
so and he uppe le canine (22 and 23). On palpa ion,
he lump was luc uan and no pain ul (Fig. 1). Tee h 22
and 23 we e i al. A cone beam compu ed omog aphy
(CBCT) was pe o med, e ealing a well-de ined, unilo-
cula adiolucen lump o 4 mm in diame e , which was
causing e osion o he es ibula co ical a ea.
Clinical and adiological aspec s led o a p esump i e
diagnosis o non-in lamma o y odon ogenic cys cau-
sing co ical pe o a ion in he maxilla, which was also
consis en wi h a gingi al cys o adul and ke a ocys
odon ogenic umo .
A cys ec omy was pe o med unde local anes hesia
wi hou conduc ing a oo canal ea men o adjacen
ee h, and he ob ained ma e ial was sen o his opa ho-
logic es ing.
A ull- hickness incision was made, p ese ing he papi-
lla o a oid u u e gum ecession (papilla-base incision),
beginning on he mesial pa o 22 and ending on he
dis al pa o 23. This incision con inues wi h a e ical
incision on he dis al a ea o 23, passing he de o mi y,
and pene a ing he mucogingi al junc ion o ha e be e
isibili y and access. A ull- hickness lap was ca e u-
lly ele a ed o a oid ea ing he lap o he cys capsule.
Once localized, he cys was emo ed ia cu e age o
he bone issue.
A e emo al, he cys was placed in 10% o malin and
was sen o a pa hological ana omy lab o his ologic
diagnosis. Subsequen ly, he lap was eposi ioned using
a 6/0 non-abso bable su u e.
The pa ien was p esc ibed an ibio ic ea men (one a-
ble o amoxicillin/cla ulanic acid 875mg/125mg e e y
8 hou s o 7 days), as well as nons e oidal an i-in lam-
ma o y d ugs (600mg o ibup o en; one able e e y 8
J Clin Exp Den . 2017;9(1):e167-71. Ke a ocys ic odon ogenic umo
e169
hou s o 3 o 5 days). The pa ien was old no o b ush
in a eas nea he incision o he i s 24 hou s, and o use
mou hwash wi h 0.12% chlo hexidine wice a day o
wo weeks. In addi ion, he pa ien was p esc ibed a so ,
cold die o a oid hea y chewing o a leas wo o h ee
days. The s i ches we e emo ed wo weeks la e .
The his opa hological indings e ealed a cys ic lesion
wi h a loose connec i e issue wall, an inne lining o
well-de ined simple squamous epi helium (4-8 cells in
hickness), basal cells a anged in a palisaded pa e n
wi h a supe icial pa ake a inized ocally co uga ed
su ace. The epi helium was ocally de ached om he
connec i e issue, and ke a in was ound inside he cys-
ic lesion. The collec ed da a es ablished a pe iphe al ke-
a ocys odon ogenic umo as he diagnosis (Fig. 2).
Pe iodic check-ups we e ca ied ou a e one mon h,
h ee mon hs, six mon hs and one yea pos -su ge y, as
well as a CBCT, which con i med no signs o ecu ence
(Fig. 3). The pa ien was asked o hei in o med con-
sen o publish hei case, gi ing his consen .
Fig. 2. A) Cys ic lesion, inne epi helial lining pa ially de ached and cys ic lumen con ains ke a in (H&E 4x). B) Simple squamous
epi helium 4-8 laye s wi h basal cells a anged in palisaded pa e n and supe icial pa ake a osis, ocally co uga ed. Capsule o med by
connec i e issue (H&E 20x). C) Simple squamous cys ic epi helium wi h palisaded basal cells and supe icial co uga ed pa ake a osis
(H&E 10x).
Fig. 3. CBCT: Sagi al sec ions a e a yea : he su gical a ea shows signs o bone healing.
Discussion
Rega ding he case’s clinical p esen a ion, he cys was
su ounded by a smoo h con ou simila o a gingi al
cys o adul , while he con ou o he pe iphe al ke a-
ocys odon ogenic umo (PKCOT) is usually i egula
and undula ing. The his ological examina ion was suc-
cess ul, as he e was e idence o “ke a in scales.” In he
majo i y o cases, his is e y di icul o achie e. The
lesion in his s udy e ealed he his ological cha ac e is-
ics o a PKCOT, including pa ake a iniza ion (8-21).
In he sys ema ic e iew ca ied ou in PubMed using
he keywo ds “pe iphe al,” “ke a ocys ic,” “odon oge-
nic,” and “ umo ,” only 22 cases o PKCOT we e ound,
se en een o which we e localized on he gum issue
(77%), h ee on he o al mucosa (14%), and wo in he
la e al acial deep egion (9%) (Table 1).
Rega ding he umo ’s loca ion, he p opo ion o maxi-
lla y KCOT, wi h espec o hose occu ed in he man-
dible, is 1 o 2 o 1 o 3. The lesion is localized in he
maxilla y ube osi y in only 10% o cases, and wi h an
e en lowe pe cen age in he canine a ea (21). PKCOT
is mos commonly ound in he mandible, since he e is
a a io o 12:7; he e a e 19 cases desc ibed in o al, 12 o
which occu ed in he maxilla and 7 in he mandible (Ta-
ble 1). Rega ding he es ibula o lingual loca ion, i is
mo e o en ound in he es ibula egion, as was he case
o he pa ien in his s udy (21). Likewise, i is impo an
o highligh he es he ic consequences in ol ed (22).
In addi ion, he umo ’s speci ic cha ac e is ics mus be
emphasized. E en hough i was o ela i ely small size
J Clin Exp Den . 2017;9(1):e167-71. Ke a ocys ic odon ogenic umo
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Au ho Yea Cases Age Gende Si e Bone a ec a-
ion*
% a ec a-
ción
Linked o
synd ome
S oelinga e al. (9) 1975 1 - - Maxilla y gingi a - - No
Buchne and Hansen (19) 1979 2 - - Buccal mucosa - - No
Buccal mucosa - - No
Dayan e al. (8) 1988 142 M Le maxilla y gingi a Yes 18% No
Chehade e al. (10) 1994 637 M Righ mandibula gingi a No 0% No
66 F Le maxilla y gingi a No 0% No
35 F Mandibula gingi a No 0% No
70 M Le mandibula gingi a No 0% No
57 F Righ maxilla y gingi a No 0% No
42 M Righ mandibula gingi a Yes 35% No
Fa dal O e al. (11) 1994 141 F Mandibula and maxilla y gingi a No 0% No
Ide e al. (12,13) 2002 238 F Le maxilla y gingi a No 0% No
46 F Righ maxilla y gingi a No 0% No
Chi e al. (14) 2005 281 F Le maxilla y gingi a Yes 10% No
64 F Le maxilla y gingi a No 0% No
Rhonda e al. (15) 2005 183 F Le maxilla y gingi a Yes 40% No
Faus ino e al. (16) 2008 157 F Le mandibula gingi a Yes 15% No
Vij e al. (17) 2011 156 M Le maxilla y gingi a Yes 30% No
G obe e al. (20) 2012 152 M Buccal mucosa No 0% No
Ling Zhu e al. (21) 2014 244 F Deep le la e al acial egion Yes 25% No
69 M Deep igh la e al acial egion Yes 8% No
Kei Sakamo o e al. (18) 2014 124 F Mandibula gingi a No 0% Yes
Cu en case 2016 132 M Le maxilla y gingi a Yes 35% No
Table. 1. Summa y o epo ed cases o pe iphe al ke a ocys odon ogenic umo (PKCOT) in he li e a u e.
*Co ical bone collapse.
(app oxima ely 4 mm), he umo had pe o a ed he
es ibula co ical a ea. The size o PKCOTs can a y,
bu he mos common dimensions a e be ween 3 and 5
mm, and hey a e a ely la ge , eaching e en 3 o 4 cm
(21). The link be ween hese umo s and Go lin-Gol z
synd ome has been desc ibed, in which case he size is
mo e commonly be ween 3 and 5 mm (18).
Con o e sy exis s o e whe he PKCOT is a locally des-
uc i e lesion wi h a high a e o ecu ence like KCOT,
o whe he i is an indolen lesion mo e simila o gingi-
J Clin Exp Den . 2017;9(1):e167-71. Ke a ocys ic odon ogenic umo
e171
al cys o adul (10-13). Ide e al. (12,13) a i med ha
he PKCOT and he KCOT we e no ex aosseous and
in aosseous a ian s wi hin he same en i y.
The ques ion o whe he o no he PKCOT is a coun-
e pa o KCOT mus be answe ed h ough exhaus i e
examina ion on a case-by-case basis. The PKCOT o he
pa ien in his s udy seems o be a ue exp ession o a
KCOT in so issue (3,5,8).
In he cases linked o Go lin-Gol z synd ome, PTCH1
mu a ions we e iden i ied, and he immunohis ochemi-
cal esul s sugges ed ha he KCOT and PKCOT le-
sions we e caused by a gene ic al e a ion in he pa ien s’
PTCH1-GLI gene (18). As all cases o Go lin-Gol z syn-
d ome ini ially ha e mu a ions in PTCH, i is logical ha
hese lesions appea .
Cu en ly, he e is no consensus on KCOT ea men due
o i s high a e o ecu ence. As he li e a u e shows,
ea men o his umo can ange om ma supializa ion
o a esec ion in-bloc, om mos conse a i e o mos
agg essi e ea men s, espec i ely (2,7,10).
PKCOT canno be comple ely compa ed o KCOT, as
he clinical and biological beha io is no necessa ily
he same o bo h. The e o e, choice o ea men will
depend on he age o he pa ien , he loca ion and size o
he umo , and whe he i is a p ima y o ecu en umo .
These ac o s will jus i y he di e ences ound in he
scien i ic li e a u e (6).
In he p esen clinical case, a comple e exe esis o he le-
sion was conduc ed wi h pos e io cu e age and a sligh
bone d ill, hus a oiding he possibili y o any emains
o he lesions emaining in he a ec ed a ea. To da e, he
pa ien p esen s no signs and symp oms o ecu ence
a e a yea .
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Con lic o In e es
The au ho s decla e ha hey ha e no con lic o in e es .