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Ciruga Bucal / Oral Surgeryy Ameloblastoma / Ameloblastoma

Ameloblastoma mandibular. Revisin de la literatura y presentacin de seis casos


Mandibular ameloblastoma. A review of the literature and presentation of six cases

Daniel Torres Lagares (1), Pedro Infante Cosso (2), Jos M Hernndez Guisado (2), Jos Luis Gutirrez Prez (3)

(1) Becario F.P.D.I.


(2) Profesor Asociado de Ciruga Bucal
(3) Profesor Titular de Ciruga Bucal. Departamento de Estomatologa. Facultad de Odontologa. Universidad de Sevilla

Correspondencia / Address:
Daniel Torres Lagares
C/ Sta M Valverde 2 3C
41008 Sevilla
Tlfno: 661 336 740
Fax 954 481 129
Indexed in:
E-mail: danieltl@us.es -Index Medicus / MEDLINE / PubMed
-EMBASE, Excerpta Medica
Recibido / Received: 1-02-2004 Aceptado / Accepted: 30-05-2004 -Indice Mdico Espaol
-IBECS

Torres-Lagares D, Infante-Cosso P, Hernndez-Guisado JM,


Gutirrez-Prez JL. Mandibular ameloblastoma. A review
of the literature and presentation of six cases. Med Oral
Patol Oral Cir Bucal 2005;10:231-8.
Medicina Oral S. L. C.I.F. B 96689336 - ISSN 1698-4447

RESUMEN ABSTRACT
El ameloblastoma es un tumor odontognico benigno de origen Ameloblastoma is a benign odontogenic tumour of epithelial
epitelial sin induccin en el tejido conectivo. En el tratamiento origin without induction in the connective tissue. In treating this
del mismo se debe valorar el tipo clnico (slido y multiqustico, type of tumour, it is important to assess the clinical type (solid,
uniqustico, perifrico), la localizacin y el tamao del tumor, multicystic, unicystic, peripheral), localisation, and size of the
as como la edad del paciente. Recientemente se han publicado tumour as well as age of the patient. Articles have recently been
algunos artculos con el fin de actualizar los conocimientos y published with the purpose of providing updated knowledge
actitudes teraputicas frente al ameloblastoma. and therapeutic approaches to ameloblastoma. We present six
Se presentan seis casos de pacientes afectos de ameloblastomas cases of patients with localised mandibular ameloblastoma who
localizados en la mandbula, tratados en los ltimos siete aos. were treated during the last seven years. We present data on
Se aportan datos acerca de su aparicin clnica, sus caracters- clinical appearance, histological characteristics, and therapeutic
ticas histolgicas, el manejo teraputico realizado que consisti approach that was used, which included excision of the lesion,
en la extirpacin de la lesin, fresado perilesional del hueso o perilesional drilling of the bone, or block resection, according
reseccin en bloque, segn el tipo de ameloblastoma. Finalmente to the type of ameloblastoma. Lastly, we analysed follow-up
analizamos el seguimiento y la aparicin de recidivas en los measures and the rate of recurrence in these patients. The aim of
pacientes presentados. this paper is to review the concepts relating to ameloblastoma
El objetivo de este artculo es revisar los conceptos respecto al that have been published recently and to assess their influence on
ameloblastoma que se han publicado recientemente y valorar the clinical attitude taken when facing this pathology, using our
su influencia en la actitud del clnico a la hora de actuar frente experience with ameloblastoma as a starting point to illustrate
a esta patologa, tomando como punto de partida para ilustrar this discussion.
est discusin la presentacin de nuestra experiencia respecto
al ameloblastoma. Key words: Odontogenic tumour, ameloblastoma, ameloblas-
toma unicystic, ameloblastoma solid, ameloblastoma multi-
Palabras clave: Tumor odontognico, ameloblastoma, amelo- cystic.
blastoma uniqustico, ameloblastoma slido, ameloblastoma
multiqustico. INTRODUCTION
Of all swellings of the oral cavity, 9% are odontogenic tumours,
INTRODUCCION and within this group, ameloblastoma accounts for 1% of lesions
El 9% de todas las tumefacciones de la cavidad oral son tumores (1-4). The WHO defines it as a locally-invasive polymorphic
odontognicos y dentro de este grupo, el ameloblastoma repre- neoplasia that often has a follicular or plexiform pattern in a
senta el 1 % de las lesiones (1-4). La O.M.S. lo define como fibrous stroma. Its behaviour has been described as being benign
una neoplasia polimrfica localmente invasiva que comnmente but locally aggressive (5,6).

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Med Oral Patol Oral Cir Bucal 2005;10:231-8. Ameloblastoma / Ameloblastoma

tiene un patrn folicular o plexiforme, en un estroma fibroso. Su It localises to the maxilla in 20% of cases, most often in the canine
comportamiento ha sido descrito como el de un tumor benigno and antral regions. In the mandible (80% of ameloblastomas),
pero localmente agresivo (5,6). 70% are located in the area of the molars or the ascending ramus,
Se localizan en el maxilar superior en un 20% de los casos, 20% in the premolar region, and 10% in the anterior region (5).
ms frecuentemente en la zona canina y antral. En la mandbula They are usually diagnosed between the fourth and fifth deca-
(80% de los ameloblastomas) el 70% se da en zona molar o rama des of life, except in the case of the unicystic variety, which is
ascendente, el 20% en regin premolar y un 10% en la regin diagnosed between the ages of 20 and 30 years, with no gender
anterior (5). Se diagnostican sobre todo entre la cuarta y quinta predilection. About 10-15% of the tumours are associated with a
dcada de vida, salvo la variedad uniqustica que se diagnostica non-erupted tooth (7). There are practically no symptoms except
entre los 20 y 30 aos, sin existir predileccin respecto al sexo. the occurrence of swelling in most cases. However, it is still infil-
De entre un 10 a un 15% de los tumores se asocian a un diente trative in character in terms of local malignant behaviour.
no erupcionado (7). La sintomatologa es prcticamente nula, Ameloblastoma is observed as a radiolucent area, which may
limitada a la provocacin de una tumefaccin en la mayora de present in three different patterns. The most common form is the
los casos, pero manteniendo un carcter infiltrante en el sentido multilocular with various cysts that are in groups or separated
de malignidad local. by osseous reinforced septa (soap bubble appearance). Another
El ameloblastoma se observa como un rea radiolcida, pudiendo image we sometimes see is a beehive pattern, this being the second
presentar tres patrones diferentes. El ms frecuente es el multilo- most common type. A third radiographic manifestation, which is
cular, con varios quistes agrupados y separados por tabiques de very important in terms of a differential diagnosis, is the unilocular
refuerzo seo (en pompas de jabn). Otras veces la imagen con form. In choosing a treatment for ameloblastomas, the clinical
que nos encontramos es en panal de abejas, siendo sta la segunda type (solid, multicystic, unicystic, peripheral), localisation, size of
en frecuencia. Una tercera imagen radiolgica, y muy importante the tumour, and age of the patient should be assessed. Resection
desde el punto de vista del diagnstico diferencial, es la forma should be as wide as possible to include healthy tissue, since
unilocular. En la eleccin del tratamiento de los ameloblastomas recurrence is fairly common with this disease (8-10).
se debe valorar el tipo clnico (slido-multiqustico, uniqustico, Articles have recently been published with the purpose of pro-
perifrico), la localizacin y el tamao del tumor, as como la edad viding updated knowledge and therapeutic approaches related
del paciente, realizndose una reseccin lo ms amplia posible to ameloblastoma. Of significance among these is the work of
para quedar en zona sana, siendo una patologa que recidiva con Reichart et al. (11), which has prompted the appearance of other
bastante facilidad (8-10). publications criticising, supporting, or completing their work.
Recientemente se han publicado algunos artculos con el fin de The aim of this article is to review the concepts that have been
actualizar los conocimientos y actitudes teraputicas frente al published recently and assess their influence on the clinical atti-
ameloblastoma. Entre ellos toma gran importancia el trabajo de tude taken when facing this pathology, using our experience with
Reichart y cols. (11) que ha provocado la aparicin de otras publi- ameloblastoma as a starting point to illustrate this discussion.
caciones criticndolo, apoyndolo o completndolo. El objetivo
de este artculo es revisar estos conceptos que se han publicado CLINICAL CASES
recientemente y valorar su influencia en la actitud del clnico a We present six cases of ameloblastoma treated during the pe-
la hora de actuar frente a esta patologa, tomando como punto riod of January 1995 to December 2001 in the Maxillofacial
de partida para ilustrar esta discusin la presentacin de nuestra Surgery Service of the Virgen del Roco Hospital in Seville
experiencia respecto al ameloblastoma. (Table 1).
All cases involved mandibular localisation. The most common-
CASOS CLINICOS ly-affected areas were the body of the mandible (four cases) with
Presentamos seis casos de ameloblastoma tratados durante el or without involvement of surrounding areas, followed by the
periodo comprendido entre enero de 1995 y diciembre de 2001 symphysis (one case), and the angle of the mandible (one case).
en el Servicio de Ciruga Maxilofacial del Hospital Virgen del In terms of gender, females outnumbered males by a 2:1 ratio.
Roco de Sevilla (Tabla 1). Mean age of the patients was 42.3 years. The youngest individual
Todos ellos fueron de localizacin mandibular, siendo las zonas was 25 years old, and the oldest was 65 years old.
ms frecuentemente afectadas el cuerpo mandibular (cuatro ca- The follow-up period was variable between patients, ranging
sos) con o sin afectacin de espacios cercanos, seguidos de la from 1 year 7 months to 5 years 11 months, with a mean of 4
snfisis (un caso) y el ngulo mandibular (un caso). Respecto al years 8 months. None of the patients has experienced recurrence
sexo, el femenino present una predileccin de dos a uno frente of the disease. The mean size of the lesion was 2.5x2.25cm, with
al masculino. La edad media de los pacientes fue de 42,3 aos, the largest being 4x3cm and the smallest 1.5x1cm.
contando el ms joven con 25 aos y el mayor 65. Case 1.- Woman, 65 years old, presenting for a tumour localised
El periodo de seguimiento fue desigual en los pacientes, yendo to the horizontal ramus of the mandible extending to the incisors
de un ao y siete meses a cinco aos y 11 meses, siendo la me- (Figure 1). The lesion was resected, preserving the base of the
dia de cuatro aos y ocho meses. Ninguno de los pacientes ha mandible, and extraction of 41, 42, and 43 was performed. An
presentado an recidiva de su patologa. El tamao medio de anatomic pathology analysis indicated the presence of a follicu-
la lesin fue de 2,5 x 2,25 cm, siendo la mayor de 4 cm x 3 cm lar and acanthomatous ameloblastoma measuring 1x0.7cm.

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Ciruga Bucal / Oral Surgeryy Ameloblastoma / Ameloblastoma

Tabla 1. Resumen de las principales caractersticas epidemiolgicas, clnicas, radiolgicas y evolutivas de los casos presentados

Tamao Subtipo
Paciente Edad Sexo Localizacin Clnica Radiologa Tratamiento Seguimiento Recidiva
(cm) Histolgico
Reseccin de la
lesin conservando
Cuerpo mandibular Folicular acan-
1 65 M 2x2 Tumefaccin Multilocular la cortical basal. 1 a. y 7 m. No
derecho tomatoso
Extraccin de 41,
42 y 43.
Escisin y Osteot-
mia perifrica.
2 59 H 3x1 Snfisis mandibular Tumefaccin Unilocular Qustico 3 a. y 6 m. No
Exodoncia de 41
y 42.
Curetaje y fresado
ngulo mandibular Qustico del hueso perilesio-
3 33 M 1 x 1,5 Tumefaccin Multilocular 4 a. y 9 m. No
derecho Acantomatoso nal. Relleno de la
cavidad.
Cuerpo y ngu-
Mixto (folicu-
4 42 M 2,7x3,5 lo mandibular Tumefaccin Unilocular Quistectoma 5 a. y 9 m. No
lar-plexiforme)
izquierdo
Exodoncia de 46,
Dolor, 47 y 48. Osteoto-
Cuerpo y ngulo Mixto (folicu-
5 25 H 2x3 tumefaccin Multilocular ma segmentaria 5 a. y 11 m. No
mandibular derecho lar-plexiforme)
y sangrado respetando cortical
basal
Curetaje y fresado
Cuerpo y ngulo
6 30 M 4x3 Tumefaccin Multilocular Qustico del hueso perilesio- 6 a. y 9 m. No
mandibular izquierdo
nal. Exodoncia 48.

Table 1. Summary of the main epidemiological, clinical, radiographic, and progressional characteristics of the cases presented in this paper.

Size Signs and Histological


Patient Age Sex Location Radiology Treatment Follow-up Recidive
(cm) symptoms subtype
Resection of the
Follicular tumor preserving
Horizontal ramus of the
1 65 M 2x2 Swelling Multilocular - Acantho- the cortical basal. 1 y. and 7 m. No
right mandible
matous Extraction of 41, 42
and 43.
Curettage and
2 59 F 3x1 Mandibular symphysis Swelling Unilocular Cystic SEPO. Extraction of 3 y. and 6 m. No
41 and 42.
Curettage y drilling
Mandible, area of the Cystic- Acan- of the perilesional
3 33 M 1 x 1,5 Swelling Multilocular 4 y. and 9 m. No
right first lower molar thomatous bone. Fill of the
cavity.
Compound
Left mandibular body
4 42 M 2,7x3,5 Swelling Unilocular (Follicular- Cystectomy 5 y. and 9 m. No
and angle
Plexiform)
Extraction of 46, 47
Pain, swe- Compound
Right mandibular body and 48. Segmental
5 25 F 2x3 lling and Multilocular (Follicular- 5 y. and 11 m. No
and angle osteotomy preserving
bled Plexiform)
the cortical basal.
Curettage y drilling
Left mandibular body
6 30 M 4x3 Swelling Multilocular Cystic of the perilesional 6 y. and 9 m. No
and angle
bone

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Med Oral Patol Oral Cir Bucal 2005;10:231-8. Ameloblastoma / Ameloblastoma

y la menor de 1,5 cm x 1 cm. Case 2.- Woman, 59 years old, with a history of chronic bron-
Caso 1.- Mujer de 65 aos que acude a consulta por tumoracin chitis, hypertension under treatment, and tonsillectomy. She was
localizada en rama mandibular horizontal, que alcanza incisivos referred by her dentist who a marked swelling in the mental
(Figura 1). Se realiz una reseccin de la lesin con conservacin region detected two months previously, and observed on radio-
de la basal mandibular y extraccin de 41, 42 y 43. El anatomo- graphic examination a unilocular radiolucent lesion extending
patlogo inform de la presencia de un ameloblastoma folicular from 35 to 45 (Figure 2). Intervention under general anaesthesia
y acantomatoso de 1 cm x 0,7 cm de tamao. was prepared, and an intraoperative biopsy was taken, which
Caso 2.-Mujer de 59 aos con antecedentes de bronquitis cr- confirmed the presence of a cystic ameloblastoma. Surgical
nica, hipertensin en tratamiento y amigdalectoma. Acude a excision of the lesion, peripheral osteotomy, and extraction of
consulta derivada por su dentista, que tras detectar hace dos 41 and 42 were performed.
meses una tumefaccin brusca mentoniana, observa en la explo- Case 3.- Woman, 33 years old, presented with a mass in the area
racin radiolgica una lesin radiolcida unilocular que abarca of the right first lower molar (Figure 3). The patient had under-
de 35 a 45 (Figura 2). Se prepara la intervencin bajo anestesia gone intervention for a residual inflammatory cyst in the same
general, realizndose una biopsia intraoperatoria que confirma area 8 years previously. A medullary and vestibular cortical bone
la presencia de un ameloblastoma de tipo qustico. Se realiza biopsy was taken at the level of 46, which yielded a diagnosis of
la escisin quirrgica de la lesin y osteotoma perifrica, as cystic acanthomatous ameloblastoma. It was elected to perform
como la exodoncia de 41 y 42. curettage of the lesion and drilling of the perilesional bone. The
Caso 3.- Mujer de 33 aos que acude a consulta por una tumo- osseous cavity was then filled with Medpore (expanded po-
racin en zona del primer molar inferior derecho (Figura 3). La rous polyethylene). A few months later, a foreign body reaction
paciente fue intervenida a propsito de un quiste inflamatorio occurred, and the rejected material had to be removed.
residual en la misma zona hace 8 aos. Se tom una biopsia de Case 4.- Woman, 42 years old, presented with a locally-expan-
hueso cortical vestibular y medular a nivel de 46, que dio como sive osteolytic lesion in the body and angle of the mandible,
resultado el diagnstico de un ameloblastoma qustico y acanto- which involved the ascending ramus and appeared unilocular
matoso. Se decidi realizar el legrado de la lesin y fresado del and ovoid (2.7x3.5 cm) and well delimited (Figure 4). It exten-
hueso perilesional. Tras lo cul se rellen la cavidad sea con ded from the beginning of the inferior dental canal to the first
Medpore (Polietileno poroso expandido). A los pocos meses molar that was present, 48, the distal root of which had been
se produjo una reaccin a cuerpo extrao y hubo que retirar el partially resorbed. A cystectomy with intraoperative biopsy was
material rechazado. performed, which confirmed the presence of ameloblastoma
Caso 4.- Mujer de 42 aos de edad, que acude a consulta pre- in the cystic wall. Extraction of 48 was also performed. Upon
sentando una lesin osteoltica y expansiva localizada en el eruption, 48 had resorbed the entire distal root of 47. There-
cuerpo y ngulo mandibular afectando a rama ascendente, en fore, because of the grave prognosis for that tooth, it was also
apariencia unilocular, de forma ovalada (2,7 cm x 3,5 cm) y bien extracted at that time.
delimitada (Figura 4). Se extiende desde el inicio del conducto Case 5.- Male, 25 years old, presented with haemorrhagic, pa-
dentario inferior hasta el primer molar presente, el 48, cuya inful swelling at the level of the right retromolar trigone. Follo-
raz distal era reabsorbida parcialmente. Se procedi a realizar wing a biopsy of the lesion, ameloblastoma was diagnosed. On
una quistectoma con biopsia intraoperatoria que confirm la radiography, a 2x3 cm lesion was observed, which involved the
presencia de un ameloblastoma en la pared qustica, as como la lower third molar (Figure 5). Treatment consisted of extraction
exodoncia del 48. En su erupcin, el 48 haba reabsorbido toda of 48 and osteotomy of the angle of the mandible, preserving the
la raz distal del 47, por lo que dado el pronstico imposible de basal cortex. The anatomic pathology analysis reported amelo-
dicho diente, tambin fue extrado en la intervencin. blastoma infiltrating and ulcerating the gingival mucosa.
Caso 5.- Varn de 25 aos de edad que acude por tumoracin Case 6.- Woman, 30 years old, smoking 30 cigarettes per day,
sangrante y dolorosa a nivel del trgono retromolar derecho. Tras drinking alcohol on weekends, and allergic to acetylsalicylic
realizar la biopsia de la lesin se diagnostica un ameloblastoma, acid presented with swelling at the level of the angle of the
que en la radiologa se observa como una lesin de 2 x 3 cm de left mandible (Figure 6). Radiographic examination revealed
tamao, en relacin con un tercer molar inferior incluido (Figura a multilocular lesion measuring 4x3 cm, involving a wisdom
5). El tratamiento efectuado consisti en la exodoncia del 48, tooth, and extending to the premolar area. With the patient under
as como la realizacin de una osteotoma del ngulo mandibu- general anaesthesia, an intraoperative biopsy was taken, which
lar respetando la cortical basal. El informe anatomopatolgico confirmed the presence of a cystic ameloblastoma. The tumour
inform acerca de un ameloblastoma que infiltraba y ulceraba was resected via the intraoral approach and the osseous cavity
la mucosa gingival. was drilled.
Caso 6.- Mujer de 30 aos de edad, fumadora de 30 cigarrillos
al da, bebedora de fin de semana y alrgica al cido acetilsalic- DISCUSSION
lico que acude a consulta por presentar una tumefaccin a nivel In our small sample, all lesions were located in the mandible.
del ngulo mandibular izquierdo (Figura 6). En la exploracin This is the most common localisation (12), while the rate of
radiolgica se aprecia una lesin multiloculada, de 4 x 3 cm de occurrence of ameloblastoma in the maxilla is lower. In terms
tamao en relacin con un cordal incluido y que se extiende hasta of age, our patients fall within the age ranges reported in the
el rea premolar. Bajo anestesia general se realiza una biopsia

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Ciruga Bucal / Oral Surgeryy Ameloblastoma / Ameloblastoma

Fig. 2. Caso 2. A: Imagen postoperatoria, en la que se observa la


reseccin realizada y la exodoncia de 41 y 42, seis meses despus de
la intervencin; B: Ortopantomografa de control, dos aos despus
de la intervencin.
Fig. 1. Caso 1. A: Lesin radiolcida a nivel del
Case 2. A: Postoperative image showing the resection and extraction
cuerpo mandibular que alcanza incisivos; B: Ra-
of 41 and 42, six months after intervention; B: Follow-up ortho-
diografa panormica postoperatoria (a los cuatro
pantomography, two years after intervention.
meses de la intervencin).
Case 1. A: Radiolucent lesion at the level of the
body of the mandible extending to the incisors;
B: Postoperative panoramic radiography (four
months after intervention).
Fig. 3. Caso 3. A: Ortopantomografa que muestra la lesin
inicial a nivel del 36-37; B: Ortopantomografa inmediata-
mente postoperatoria; C: Ortopantomografa de control en
la que se observa la formacin de hueso (cuatro aos tras la
intervencin).
Case 3. A: Orthopantomography showing the initial lesion at
the level of 36-37; B: Immediate postoperative orthopanto-
mography; C: Follow-up orthopantomography showing bone
formation, four years after intervention.

Fig. 4. Caso 4. A: Lesin osteoltica y expansiva a nivel del ngulo mandibular


izquierdo; B: TAC de la misma lesin (preoperatorio); C: TAC con constraste
intravenoso, aproximadamente al mismo nivel que el corte anterior; D: Ortopanto-
mografa postoperatoria, a los seis meses de la intervencin.
Case 4. A: Expansive osteolytic lesion at the level of the angle of the left mandible;
B: CT image of the same lesion (preoperative); C: CT image with intravenous
contrast, at approximately the same level as the previous section; D: Postoperative
orthopantomography, six months after intervention.

Fig. 5. Caso 5. A: Lesin inicial en relacin con el tercer molar


inferior derecho incluido; B: Ortopantomografa inmedia-
Fig. 6. Caso 6. A: Lesin inicial, mostrando una aparente multilocularidad y tamente postoperatoria; C: Ortopantomografa de control, a
multilobularidad; B: Ortopantomografa inmediatamente postoperatoria; C: los dos aos de la intervencin. Obsrvese el remodelado del
Ortopantomografa de control, dos aos despus de la intervencin; D: Recons- hueso alveolar.
truccin 3D de la mandbula, dos aos despus de la intervencin. Case 5. A: Initial lesion involving the right lower third molar;
Case 6. A: Initial lesion showing apparent multilocularity and multilobularity; B: Immediate postoperative orthopantomography; C: Follow-up
B: Immediate postoperative orthopantomography; C: Follow-up orthopanto- orthopantomography, two years after intervention. Remodelling
mography, two years after intervention; D: 3-D reconstruction of the mandible, of alveolar bone is visible.
two years after intervention.

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intraoperatoria que confirma la presencia de un ameloblastoma literature as standard for this type of pathology (13). Most au-
qustico, efectundose la reseccin de la lesin tumoral va in- thors do not report differences in gender, though the majority
traoral y el fresado de la cavidad sea. of these patients may be male (14,15). In terms of clinical pre-
sentation, typical findings with this disease include swelling in
DISCUSION all cases with the occurrence of other incidents (haemorrhage,
En nuestra pequea muestra, todas las lesiones asientan en la pain, etc.) (15).
mandbula. sta es la localizacin ms frecuente (12), mientras Although in some of our cases, the follow-up period was short,
que la frecuencia de aparicin de un ameloblastoma a nivel in four of our patients, it lasted up to five years or more, and
del maxilar superior es menor. Respecto a la edad, nuestros in only one case was it less than three years. For this reason,
pacientes se encuentran dentro de los lmites que la literatura we believe the data on recurrence rates are valid and should be
marca como usuales para la aparicin de esta patologa (13). La considered. Three types of ameloblastoma have classically been
mayor parte de los autores no apuntan diferencias respecto al described: solid or multicystic, unicystic, and peripheral. It ap-
sexo, aunque la mayor parte de nuestros pacientes sean varones pears that Melrose was correct in the proposal to group the two
(14,15). Respecto a la clnica encontrada es la tpica en este tipo forms from the first group under the designation of infiltrative
de patologa: tumefaccin en todos los casos con la aparicin ameloblastoma (16), since that is their primary characteristic,
de otros accidentes (sangrado, dolor, etc.) (15). which has determined the need for resection of this type of
Pese a que en algunos de nuestros casos el tiempo de seguimiento ameloblastoma with a safety margin of 1-2cm in width (17).
es escaso, en cuatro de nuestros pacientes es cercano o superior The peripheral ameloblastoma is confined within the gums or
a cinco aos, y en tan slo uno es menor a tres aos, por lo que alveolar mucosa, and is therefore not the subject of this discus-
creemos que los datos acerca de la aparicin o no de recidivas sion since none of our cases were of this type. Unicystic amelo-
son vlidos y deben ser tenidos en cuenta. Clsicamente se han blastoma is a form that was described in 1977 by Robinson and
diferenciado tres tipos de ameloblastomas: el slido o multiqus- Martnez and has been characterised (18) by its histology and
tico, el uniqustico y el perifrico. Parece correcta la proposicin its presentation at an earlier age than infiltrative ameloblastoma
que realiza Melrose de englobar a las dos formas del primer (20-30 vs. 30-50 years) (19) as well as its capacity for recurrence
grupo bajo el epgrafe de ameloblastoma infiltrante (16), dado in the face of conservative treatment, which is lower than the
que es su principal caracterstica, lo que ha condicionado unas latter (10-20% vs. 45%) (8-10,20).
resecciones de este tipo de ameloblastomas con un margen de But even within the group of unicystic ameloblastomas, there
seguridad de entre 1 y 2 cm de amplitud (17). are exceptions. Melrose proposed three possible origins of the
El ameloblastoma perifrico es aqul que est confinado en la unicystic ameloblastomas. When it originates from an infiltrative
enca o la mucosa alveolar y por lo tanto no es objeto de esta ameloblastoma, treatment should be more aggressive (16,21).
discusin ya que ninguno de los casos presentados es de este tipo. Other authors have developed distinct classifications at the his-
El ameloblastoma uniqustico es una variedad descrita en 1977 tological level, concluding that when a unicystic ameloblastoma
por Robinson y Martnez y que se ha caracterizado (18), adems displays intramural localisation of growth, treatment should be
de por su histologa, por presentarse a una edad ms temprana equally aggressive as in the case of infiltrative ameloblasto-
que el ameloblastoma infiltrante (20-30 aos frente a 30-50) (19) ma (13,18,21). It cannot currently be concluded that these
y una capacidad de recidiva, frente al tratamiento conservador, two groups are identical, although Melrose has proposed the
menor que la de ste (10-20% frente al 45%) (8-10,20). revision of the concept of the unicystic ameloblastoma, speci-
Pero incluso dentro del ameloblastoma uniqustico deben ha- fically to attempt to distinguish forms with different biological
cerse excepciones. Melrose propone tres posibles origenes del behaviour (16).
ameloblastoma uniqustico. En el caso de que se originase a To this end, attempts have been made to use different markers
partir de un ameloblastoma infiltrante el tratamiento debera ser to differentiate the type of ameloblastoma, though there have
ms agresivo (16,21). Otros autores han desarrollado distintas been no encouraging results as of yet (22,23). It is therefore of
clasificaciones a nivel histolgico, llegando a la conclusin de vital importance to consider the differentiating characteristics
que si en el ameloblastoma uniqustico se localiza un crecimien- of these entities, particularly in terms of patient age and radio-
to intramural el tratamiento debe ser igual de agresivo que si graphic appearance of the lesion (24).
estuviesemos ante un ameloblastoma infiltrante (13,18,21). Es- According to recent articles published on this subject (15,25),
tablecer identidad entre ambos grupos en la actualidad no puede unilocular or multilocular images may be seen with both types
defenderse, aunque ya Melrose propuso la revisin del concepto of ameloblastoma. However, with unicystic ameloblastomas, it
de ameloblastoma uniqustco, percisamente para intentar sepa- is more common to see a unilocular image, while a multilocular
rar formas con comportamientos biolgicos diferentes (16). image is more common with the infiltrative form. There are opi-
En este sentido se ha intentado el uso de distintos marcadores nions that contradict this claim (24) and suggest that unicystic
que diferencien el tipo de ameloblastoma, aunque sin resultados ameloblastomas presenting with multilocularity would be due
esperanzadores por el momento (22,23). Por tanto es de vital to lesions with imprecise margins, an idea that was previously
importancia considerar las caractersticas diferenciales entre presented by Ackerman et al. (21). We do not support this theory
ambas entidades, sobre todo las referidas a la edad del paciente since two of the three cystic ameloblastomas we presented occu-
y al aspecto radiolgico de la lesin (24). rred in patients with multilocular images with fairly well-defined
Segn recientes artculos publicados sobre el tema (15,25), las borders (cases 3 and 6).

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Ciruga Bucal / Oral Surgeryy Ameloblastoma / Ameloblastoma

imgenes unilocular o multilocular son posibles en ambos tipos However, in the case of a unilocular radiographic image, we
de ameloblastoma, slo que en los ameloblastomas uniqusti- advocate conservative treatment (cases 2 and 4), without the
cos es ms frecuente encontrar una imagen unilocular y en el risk of dealing with an infiltrative ameloblastoma, as occurred
infiltrante es ms frecuente la multilocular. Existen opiniones in case 4. In the case of a multilocular image, we should seek
contrarias a esta afirmacin (24) y apuntan que las imgenes the clearest possible evidence of osseous trabeculation, which
de ameloblastomas uniqusticos que presentan multilocularidad can orient us toward infiltrative ameloblastoma. We should also
seran debidas a lesiones con mrgenes imprecisos, aspecto ya consider localisation and extent of the lesion. In case 1, we saw a
sealado anteriormente por Ackerman y cols (21). No apoyamos multilocular image with the appearance of trabeculation, and the
esta ultima idea, puesto que dos de los tres ameloblastomas patient was treated by means of resection preserving the base. In
qusticos que presentamos se dan en pacientes con imagen de case 6, in which osseous trabeculation could not be compatible
multilocularidad con bordes bastante bien establecidos (casos with a single space-occupying lesion and the extent was greater
3 y 6). and more critical, we opted for conservative treatment. A cystic
No obstante, ante una imagen radiolgica unilocular abogamos ameloblastoma was later diagnosed.
por un tratamiento conservador (casos 2 y 4), sin perjuicio de This approach of avoiding aggressive treatment when possible
estar ante un ameloblastoma infiltrante, como ocurre en el even at the risk of increasing the likelihood of recurrence is
caso 4. Ante una imagen de multilocularidad, debemos buscar reflected in the literature, particularly in growing patients in
la evidencia ms clara posible de trabeculacin sea, lo que which maintenance of continuity of the mandibular base allows
puede orientarnos hacia un ameloblastoma infiltrante. Tambin for correct development of the same (26,27). Except in cases that
debemos considerar la localizacin de la lesin y su extensin. are exceptions to this type, we advocate resection of the lesion
En el caso 1 vemos una imagen multilocular con apariencia with safety margins of 2cm, as we performed in case 5.
de trabeculacin que fue tratada mediante reseccin que con- In terms of extraction of teeth near or adjacent to the lesion,
servaba la basal. En el caso 6, en que la trabeculacin sea no extraction is indicated for teeth that are within the safety mar-
poda ser compatible con una sola lesin ocupante y en que la gins of resection around the lesion. The conservation of teeth
extensin era mayor y ms crtica, se opt por un tratamiento in the alveolar bone surrounding the lesion presents a risk for
conservador, diagnosticndose posteriormente un ameloblasto- later recurrence. Furthermore, it is obvious that a peripheral
ma de tipo qustico. osteotomy, however small it may be, could affect the viability
Este tipo de actuaciones que intentan evitar en lo posible una of adjacent teeth. For this reason, the prognosis for each tooth
accin agresiva, an a costa de aumentar la posibilidad de re- should be analysed for each case. Following these criteria, the
cidivas, est reflejada en la literatura y sobre todo en pacientes teeth described in clinical cases 2, 3, 4, and 6 were extracted.
en crecimiento en el que el mantenimiento de la continuidad de Moreover, extraction of the lower second molar in clinical case
la basal mandibular permite un correcto desarrollo de la misma 4 was indicated due to its grave prognosis.
(26,27). En ausencia de consideraciones excepcionales de este In situations where circumstances point to a tumour with an
tipo, abogamos por la reseccin de la lesin con mrgenes de increased infiltrative potential, as in case 5 (multicystic type,
seguridad de 2 cm, como realizamos en el caso 5. mucosal erosion, significant clinical signs), more extensive
Respecto a la extraccin de dientes cercanos o adyacentes a la le- extraction would be indicated.
sin, est indicada la extraccin de los dientes que se encuentren Beyond surgery, there are other alternative therapies for ame-
incluidos en el margen de reseccin de seguridad alrededor de la loblastoma, such as radiation therapy. There is published evi-
lesin. el hecho de conservar los dientes en el hueso alveolar que dence on the usefulness of radiation therapy in the treatment
rodea la lesin supone un riesgo para la aparicin de posteriores of extraosseous ameloblastomas (28-30). However, the tumour
recidivas. Por otra parte, es obvio que la osteotoma perifrica, was never controlled with this method of therapy. It has not
por pequea que sea, podra afectar la viabilidad de los dientes been possible to provoke the occurrence of ameloblastoma in
adayacentes, por lo que se impone analizar individualmente el animals, and the closest available model is the so-called acan-
pronstico de cada diente segn el caso. Siguiendo este criterio thomatous epulis in dogs (31,32), which becomes malignant
se extrajeron los dientes enumerados en los casos clnicos 2, 3, 4 after application of radiation therapy. For this reason, the use of
y 6. Por otra parte, la extraccin del segundo molar inferior en el this method is unacceptable in the treatment of ameloblastoma
caso clnico 4 estaba indicada dado su pronstico imposible. in humans (33).
En situaciones en que existan circunstancias que nos hagan pen-
sar en un potencial infiltrante aumentado del tumor, como en el
caso 5 (tipo multiqustico, erosin mucosa, clnica importante)
estara indicado realizar exodoncias ms amplias.
Dejando atrs el tratamiento quirrgico del ameloblastoma, exis-
ten otras alternativas, como es la radioterapia. Existen evidencias
publicadas de la utilidad de la radioterapia en el tratamiento de
los ameloblastomas extraseos (28-30). Sin embargo nunca el
tumor pudo ser controlado con esta teraputica. No se ha podido
provocar la aparicin de ameloblastomas en animales y el mo-
delo ms cercano es el denominado epulis acantomatoso que se

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Med Oral Patol Oral Cir Bucal 2005;10:231-8. Ameloblastoma / Ameloblastoma

da en los perros (31,32), el cual se maligniza tras la aplicacin


de radioterapia, razn por la cual su uso es inadmisible en el
tratamiento del ameloblastoma en humanos (33).

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