Large ameloblastic fibro-odontoma in a 7-year-old girl with analysis of 108 cases

© 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 Access his a icle online Websi e: www.amsjou nal.com DOI: 10.4103/2231-0746.186132 Quick Response Code: Thisis anopen accessa icle dis ibu edunde  he e ms o  he C ea i e CommonsA ibu ion-NonComme cial-Sha eAlike3.0License, whichallows o he s o emix, weak,andbuildupon hewo knon-comme cially,aslongas he au ho isc edi edand henewc ea ionsa elicensedunde  heiden ical e ms. Fo ep in s con ac : ep in [email protected] Ci e his a icle as:Ki ja ainenA,Tuo inenV,Sándo  GK.La ge ameloblas ic ib o-odon omaina7-yea -oldgi lwi hanalysiso 108cases. AnnMaxillo acSu g2016;6:15-20. O iginal A icle - E alua i e S udy [Downloaded ee om h p://www.amsjou nal.com on Tuesday, Decembe 20, 2016, IP: 153.1.49.17] 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 [Downloaded ee om h p://www.amsjou nal.com on Tuesday, Decembe 20, 2016, IP: 153.1.49.17] 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... [Downloaded ee om h p://www.amsjou nal.com on Tuesday, Decembe 20, 2016, IP: 153.1.49.17] 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 [Downloaded ee om h p://www.amsjou nal.com on Tuesday, Decembe 20, 2016, IP: 153.1.49.17] 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. 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