© 2016 Annals o Maxillo acial Su ge y | Published by Wol e s Kluwe - Medknow 15
La ge ameloblas ic ib o‑odon oma in a 7‑yea ‑old gi l
wi h analysis o 108 cases
An i Ki ja ainen, Veikko Tuo inen, Geo ge K. Sándo 1,2
Depa men o O al and Maxillo acial Su ge y, Jy äskylä Cen al Hospi al, Jy äskylä, 1Depa men o O al
and Maxillo acial Su ge y, Medical Resea ch Cen e , Oulu Uni e si y Hospi al, Ins i u e o Den is y,
Uni e si y o Oulu, Oulu, 2BioMediTech, Ins i u e o Bioscience and Technology, Uni e si y o
Tampe e, Tampe e, Finland
Add ess o co espondence:
P o . Geo ge K. Sándo , Uni e si y o Oulu, Aapis ie 5A, O ice No: 2126B, 90014 Oulu, Finland.
E‑mail: geo ge.s[email p o ec ed]
INTRODUCTION
Ameloblas ic ib o‑odon oma (AFO) is one o he mixed
adiolucen and adiopaque odon ogenic umo s. I is ela i ely
a e. S udies indica e ha AFO is seen anging om 0.3%
o 1.7% o o al pa hology biopsy specimens submi ed as
possible odon ogenic umo s.[1,2] AFO was o iginally e med
ameloblas ic odon oma be o e he cu en nomencla u e. Only
a ew s udies exis ega ding he cha ac e is ics o AFOs as mos
case epo s ake he o m o single pa ien publica ions.[3] This
s udy aimed o e iew cases o AFO epo ed consecu i ely
in he li e a u e be ween he yea s 1967 and 2015 wi h he
addi ion o a new case.
Case epo
An o he wise heal hy 7‑yea ‑old gi l was e e ed by he den is
because o swelling o he le side o he mandible. An asymme ic
acial swelling was ob ious on ex ao al examina ion [Figu e 1].
The e was no his o y o auma o local in ec ion. In ao al
examina ion e ealed no mal mucosa o e lying he lesion
and he absence o he le mandibula i s and second mola
ee h [Figu e 2]. A dis inc i e ha d bulge was palpable in he
es ibule o he le mandible mola egion. The mou h opening
was no mal and he e was no pain o o he symp oms apa om
he swelling. Pano amic adiog aphy [Figu e 3] and cone‑beam
compu ed omog aphy scan [Figu es 4 and 5] bo h e ealed a la ge
well‑de ined adiolucen a ea ha con ained se e al adiopaque
s uc u es o a ying sizes and shapes. The lesion ex ended om
he i s mola o he co onoid p ocess and he condyla neck.
The bo de s o he lesion we e well ci cumsc ibed and he lesion
had displaced he de eloping second mola oo h bud down o
he in e io bo de o he mandible.
Backg ound: Odon ogenic umo s such as ameloblas ic ib o‑odon oma (AFO) a e a e condi ions in child en and a e o en
asymp oma ic. AFOs a e ound by ou ine clinical and adiological examina ion o when hey cause ob ious in a‑ o ex a‑o al
swelling. Ma e ials and Me hods: A case o an AFO in a 7‑yea ‑old gi l is desc ibed, and 107 cases om he li e a u e and his
epo a e analyzed. Resul s: The o al o 108 cases e ealed he a e age age a p esen a ion o AFO o be 6.3 yea s in boys and
9.6 yea s in gi ls. The e was a sligh male p edilec ion and AFO lesions mos o en occu ed in he pos e io mandible. AFO was
almos always associa ed wi h an une up ed oo h o ee h. Conclusions: While he ecu ence a e o AFO was ound o be
5.5%, long‑ e m pos ope a i e clinical and adiological ollow‑up is ad ised o ensu e no u u e signs o agg essi e ecu ence.
Keywo ds: Ameloblas ic ib o‑odon oma, benign umo s, mixed odon ogenic umo s
ABSTRACT
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ameloblas ic ib o-odon omaina7-yea -oldgi lwi hanalysiso 108cases.
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O iginal A icle - E alua i e S udy
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Ki ja ainen, e al.: 108 cases o ameloblas ic ib o‑odon oma
Annals o Maxillo acial Su ge y | Janua y - June 2016 | Volume 6 | Issue 1
16
Figu e 1: Ex ao al swelling o he le side o he mandible
Figu e 2: Missing o absen mola s o he le side o he mandible
Figu e 3: P eope a i e pano amic adiog aph showing la ge mixed
adiolucen and adiopaque lesion o he le amus and body o mandible
Figu e 4: P eope a i e cone-beam compu ed omog aphy scan e ealing
la ge mixed lesion o he le amus and body o mandible
Figu e 5: P eope a i e cone-beam compu ed omog aphy scan co onal
iew o mixed lesion
Figu e 6: Resec ed specimen deli e ed using an in ao al app oach
Figu e 7: Ameloblas ic ib o-odon oma wi h odon ogenic epi helium (1)
and odon ogenic s oma (2) (H and E, ×20)
Figu e 8: Ameloblas ic ib o odon oma exhibi ing a eas o odon ogenic
epi helium (1) and (2) (H and E, ×40)
Figu e 9: Pano amic adiog aph aken 6 mon hs ollowing su ge y wi h
healing o in aosseous lesion o he le amus and body o mandible
The lesion was enuclea ed using an in ao al app oach unde gene al
anes hesia. Du ing he same p ocedu e, oo h numbe s 75 and 37
we e also ex ac ed. The umo measu ed 10 cm × 4 cm × 4 cm in
size [Figu e 6] and was submi ed o his opa hological examina ion.
The samples we e s ained wi h hema oxylin and eosin. A la ge
numbe o epi helial s ands and islands o odon ogenic epi helium
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Ki ja ainen, e al.: 108 cases o ameloblas ic ib o‑odon oma
Annals o Maxillo acial Su ge y | Janua y - June 2016 | Volume 6 | Issue 1 17
Table 1: Summa y o epo ed cases o ameloblas ic ib o‑odon oma
Fi s au ho Yea o
publica ion
Numbe
o cases
Osseous
loca ion
A ea Une up ed
ee h (cases)
Sex A e age
age
Recu ance
epo ed
Ki ja ainen 2015 1 Mand=1 Mol Yes Female=1 Female=7 No
Gan ala 2015[2] 1 Mand=1 Mol Yes Female=1 Female=11 No
Su ej Kuma 2014[4] 1 Max=1 Mol Yes Female=1 Female=10 No
Banihashem Rad 2014[5] 1 Max=1 Mol Yes Female=1 Female=11 No
Lin 2014[6] 1 Max=1 Inc No Female=1 Female=3 No
S eena h 2014[7] 1 Mand=1 Inc Yes Female=1 Female=5 No
Nelson 2014[8] 1 Max=1 Mol Yes Female=1 Female=15 No
Buchne 2013[3] 10 Max=4,
Mand=6
Inciso =1, P emol=1,
Mol=5, Mul i=3
Yes=9, No=1 Female=4,
male=6
Female=14,
male=15
U=10
de Souza Tolen ino 2010[9] 1 Mand=1 Mol Yes Female=1 Female=11 No
Wewel 2010[10] 1 Mand=1 Inc Yes Male=1 Male=2 No
Ca alcan e 2009[11] 1 Mand=1 Mol Yes Male=1 Male=8 No
De Riu 2009[12] 1 Max=1 Mol Yes Male=1 Male=26 No
Nascimen o 2009[13] 1 Mand=1 Mul i Yes Male=1 Male=11 No
Zouha y 2008[14] 1 Max=1 Mul i Yes Female=1 Female=7 No
Hegde 2008[15] 1 Max=1 Mul i Yes Female=1 Female=12 U
Dolanmaz 2008[16] 1 Max=1 Mul i Yes Female=1 Female=9 No
Reis 2007[17] 1 Mand=1 Mol Yes Female=1 Female=6 No
Gyulai-Gaál 2007[18] 1 Mand=1 Mul i Yes Male=1 Male=9 No
Chang 2007[19] 1 Mand=1 Mol Yes Female=1 Female=16 U
Oghli 2007[20] 1 Mand=1 Mul i Yes Male=1 Male=3,5 No
Sil a 2006[21] 1 Mand=1 Mol Yes Male=1 Male=14 No
Hu 2006[22] 1 Max=1 Mul i Yes Female=1 Female=17 No
Soa es 2006[23] 1 Max=1 Mul i Yes Male=1 Male=4 U
Chen 2005[24] 7 Max=2,
Mand=5
Inc=1, Mol=4, Mul i=2 Yes Female=3,
male=4
Male=10,
emale=10
Yes=3, no=2,
U=2
Si apa hasundha am 2005[25] 1 Mand=1 Mul i Yes Male=1 Male=17 U
Reicha 2004[26] 2 Mand=2 Mol=2 Yes, No Male=2 Male=10 No=1, U=1
Dhanu hai 2004[27] 1 Max=1 Inc Yes Female=1 Female=1 No
Alde son 2004[28] 1 Mand=1 Mol Yes Male=1 Male=11 U
Chang 2002[29] 1 Mand=1 Mol Yes Female=1 Female=26 No
Mosqueda-Taylo 2002[30] 3 Max=2,
Mand=1
Mul i=3 Yes Female=1,
male=2
Female=25,
male=12
U=1, No=2
F ied ich 2001[31] 1 Mand=1 Mol Yes Male=1 Male=8 U
Yagishi a 2001[32] 1 Mand=1 Mul i No Male=1 Male=24 U
al-Sebaei 2001[33] 1 Max=1 Mul i Yes Male=1 Male=11 U
Flai z 2001[34] 1 Mand=1 Inc Yes Male=1 Male=2,5 U
S einbe g 2001[35] 1 Max=1 Mul i Yes Male=1 Male=6 U
Rao 1999[36] 2 Mand=2 Mul i Yes Male=2 Male=10,5 U=2
Fu s 1999[37] 1 Mand=1 Mol Yes Male=1 Male=7 Yes
Sa i ha 1998[38] 1 Mand=1 Mul i Yes Male=1 Male=5 No
Fa ia 1997[39] 2 Max=1
Mand=1
Mul i=2 U Male=2 Male=3,5 No=2
Oze 1997[40] 1 Max=1 Mul i Yes Female=1 Female=7 No
Ki ano 1994[41] 1 Mand=1 Mol Yes Female=1 Female=9 No
Bake 1993[42] 1 Max=1 Inc Yes Male=1 Male=0,75 No
Oku a 1992[43] 2 Mand=1,
Max=1
Mol=2 Yes Male=1,
emale=1
Male=7,
emale=12
No=2
Pie e 1990[44] 1 Max=1 Mul i Yes Male=1 Male=7 U
Glickman 1989[45] 1 Mand=1 Mol Yes Female=1 Female=15 U
Wa nock 1989[46] 1 Mand=1 Mul i Yes Male=1 Male=11 U
Hansen 1988[47] 8 Mand=4,
Max=4
U=8 U Male=6,
emale=2
Male=11,5,
emale=8
No=4, U=4
Takeda 1988[48] 1 Max=1 Mol Yes Female=1 Female=11 No
Hawkins 1986[49] 1 Max=1 Mol Yes Female=1 Female=6,5 U
Sole 1986[50] 1 Mand=1 Mol Yes Male=1 Male=12 U
Reich 1984[51] 1 Mand=1 Mol Yes Female=1 Female=11 No
Anne o h 1982[52] 1 Max=1 Inc Yes Male=1 Male=7 No
Hu 1982[53] 1 Mand=1 Mol Yes Female=1 Female=11 No
Daley 1982[54] 1 Max=1 Mol Yes Female=1 Female=4 U
Cu an 1980[55] 1 Mand=1 Mul i Yes Male=1 Male=15 No
Be nho 1979[56] 1 Max=1 Mol Yes Female=1 Female=14 No
Gelle 1978[57] 1 Max=1 Mol Yes Male=1 Female=15 No
Howell 1977[58] 2 Mand=2 Mul i=2 U Male=1,
emale=1
Male=36,
emale=18
Yes=2
Album 1977[59] 1 Mand=1 Mul i Yes Male=1 Male=5 No
Con d...
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Ki ja ainen, e al.: 108 cases o ameloblas ic ib o‑odon oma
Annals o Maxillo acial Su ge y | Janua y - June 2016 | Volume 6 | Issue 1
18
we e ound. The ha d issue componen consis ed o den in and
enamel. His opa hological examina ion [Figu es 7 and 8] con i med
he clinical and adiological diagnosis o AFO. Clinical and
adiological ollow‑up [Figu e 9] was con inued o e 24 mon hs and
he pos ope a i e cou se was une en ul wi h no ecu ences o da e.
MATERIALS AND METHODS
A sea ch was conduc ed using Medline and Scopus da abases
using he sea ch wo ds AFO and odon ogenic umo s. This
yielded a o al o 1254 hi s which included many lesions o he
han AFO, such as ameloblas ic ib oma (AF), ameloblas ic
ib oden inoma, ameloblas ic ib osa coma, and odon oma. The
sea ch was es ic ed o AFO and epo s including he o he
diagnoses we e emo ed. The c i e ia o inclusion in his e iew
we e epo s desc ibing adiog aphic ea u es consis en wi h
AFO, his ologic ea u es consis en wi h AFO, and minimum
ollow‑up o 6 mon hs ollowing emo al o he lesion. Any epo
o s udy ailing o men ion any o he abo e inclusion c i e ia
was excluded. This yielded a o al o 107 cases which oge he
wi h he newly epo ed case o he 7‑yea ‑old gi l comp ising
108 cases o ming he da abase o his e iew.
RESULTS
A summa y o he indings o AFO epo s is p esen ed in
Table 1. AFO was ound o be a benign umo o child en wi h
he a e age age a p esen a ion being 6.3 yea s in males and
9.6 yea s in emales. AFO had a emale: male a io o 1:1.62.
Typically, AFO lesions we e loca ed in he pos e io egion o he
mandible in 60% o cases. Almos all lesions, 94 o 106 (89%),
we e associa ed wi h he c own o an une up ed oo h o ee h.
Clinically, AFO was usually painless and was cha ac e ized by
asymme ic swelling o he ace and delayed oo h e up ion.
Radiological ea u es o AFO included a cys ‑like lesion wi h
adiopaque a eas esembling a de eloping odon oma. The
ecu ence a e in his e iew o 108 cases was only 6 ecu en
cases o 5.5% [Tables 1 and 2].
DISCUSSION
In de ining AFO, he Wo ld Heal h O ganiza ion lis s i as
a a e odon ogenic umo wi h he his ologic ea u es o AF
in conjunc ion wi h he p esence o den in and enamel.[73]
His opa hologically, he umo is composed o bo h so and
ha d issues. The so issue componen is composed o epi helial
s ands and small islands o odon ogenic epi helium associa ed
wi h a p imi i e appea ing myxoid connec i e issue which
esembles he den al papilla [Figu es 7 and 8]. The ha d issue
componen consis s o oci o enamel and den in.[74]
S udies ha e shown ha AFO is usually no agg essi e and can be
ea ed su gically wi hou emo al o he adjacen ee h.[43,68,71,70,75]
A ew s udies sugges ha AFO can beha e agg essi ely wi h
mul iple ecu ences.[24,76] The e had been sugges ions ha a
p ima ily benign AFO can unde go malignan ans o ma ion
o ameloblas ic ib osa coma.[58,77‑79] The e o e, i is impo an o
con inue he ollow‑up o AFO lesions o se e al yea s.
CONCLUSIONS
AFO is a a e benign mixed odon ogenic umo . No mally, AFO
is ound a he a e age age o 9 yea s in emales and 6 yea s in
males. The p ognosis is good. The co ec diagnosis is based
on clinical, adiological, and his opa hological indings. AF
and complex odon oma mus be conside ed in he di e en ial
Table 2: Cha ac e is ics o 108 ameloblas ic
ib o‑odon oma lesions
Cha ac e is ic Ra io
Female o male a io 1:1,65
A e age age (yea s) Female: 9.6
Male: 6.3
Mandible e sus maxilla (%) 65 (59.43) e sus 43 (40.57)
Mola egion (%) Mola : 47 (44.34)
Mola + mul iple si es: 85 (80.19)
Une up ed ee h (%) 94 (88.68)
Recu ence (%) 6 (5.57)
He d 1977[60] 1 Mand=1 Mol Yes Female=1 Female=11 No
Eda 1977[61] 1 Max=1 Mul i Yes Female=1 Female=22 No
C an 1976[62] 1 Mand=1 Mol Yes Male=1 Male=9 U
Hanna 1976[63] 1 Mand=1 Mul i Yes Male=1 Male=11 No
Mille 1976[64] 7 Mand=4,
Max=3
Mol=4, Mul i=3 Yes=4, U=3 Male=5,
emale=2
Male=11,
emale=12
No=7
Mee ko e 1974[65] 1 Mand=1 Mol Yes Male=1 Male=9 No
Sande s 1974[66] 1 Mand=1 Mol Yes Male=1 Male=19 No
Wo ley 1972[67] 1 Mand=1 Mul i Yes Male=1 Male=11 U
O`B ien 1971[68] 1 Max=1 U U Female=1 Female=2,5 U
Du a 1970[69] 1 Max=1 Mul i Yes Male=1 Male=8 No
Jacobsohn 1968[70] 1 Mand=1 P emol No Male=1 Male=15 No
Hamne 1968[71] 2 Mand=2 Mol=2 Yes=2 Male=2 Male=7 No=2
Olech 1967[72] 1 Max=1 Inc Yes Female=1 Female=5 No
To al 108 Max=43,
Mand=65
Inc=10, P emol=2,
Mol=49, Mul i=38, U=9
Yes=96,
no=5, U=7
Male=66,
emale=42
Female=9,6,
male=6,3
No=62,
Yes=6, U=40
Max=Maxilla; Mand=Mandible; Inc=Inciso ; P emol=P emola ; Mol=Mola ; Mul i=Mul iple si es; U=Unknown. Nume ical supe sc ip nex o yea o
publica ion indica es ci a ion numbe o epo in e e ences sec ion o manusc ip
Table 1: Con d...
Fi s au ho Yea o
publica ion
Numbe
o cases
Osseous
loca ion
A ea Une up ed
ee h (cases)
Sex A e age
age
Recu ance
epo ed
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Ki ja ainen, e al.: 108 cases o ameloblas ic ib o‑odon oma
Annals o Maxillo acial Su ge y | Janua y - June 2016 | Volume 6 | Issue 1 19
diagnosis. The ea men o choice is o emo e umo su gically
usually by enuclea ion.
In his newly epo ed case, he AFO umo was enuclea ed and
he a ea o he le pos e io mandible healed une en ully wi hou
complica ions. Clinical and adiological ollow‑up has con inued
o 24 mon hs wi h no signs o ecu ence. Follow‑up mus be
con inued on a egula basis o se e al yea s. The pa ien in his
case epo is now ecei ing o hodon ic ea men and emains
unde igilan su gical ollow‑up.
Financial suppo and sponso ship
Nil.
Con lic s o in e es
The e a e no con lic s o in e es .
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Ki ja ainen, e al.: 108 cases o ameloblas ic ib o‑odon oma
Annals o Maxillo acial Su ge y | Janua y - June 2016 | Volume 6 | Issue 1
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