Australian Dental Journal 1997;42:(5):315-8
Adenomatoid odontogenic tumour
(adenoameloblastoma). Case report and review of the
Ertunç Dayı, DDS, PhD*
u u u
Geleng¨ l G¨ rb¨ z, DDS†
O. Murat Bilge, DDS, PhD‡
M. Akif Çiftcioˇ lu, MD, PhD§
Abstract The tumour affects females more than males in
An adenomatoid tumour was found in the anterior almost a two to one ratio.2-5 The maxilla is involved
maxillary region of a 15 year old female patient. nearly twice as frequently as the mandible.2-5,9,10
Two impacted teeth were found in the tumour. The Unerupted permanent teeth were associated with
lateral incisor found in the tumour was dilacerated, this lesion in one-third of the cases.2-5 In a few cases,
and the roots of the first premolar were resorbed. A more than one unerupted tooth was associated with
review of the English literature indicated that 294
similar cases have been reported. the tumour.4,5
Key words: Adenomatoid odontogenic tumour, case
The cuspid is the tooth most commonly associated
report. with the adenomatoid odontogenic tumour.4,5,8,11
(Received for publication March 1994. Revised
Three-quarters of the tumours involved the anterior
December 1994. Accepted January 1995.) aspect of the jaws, particularly the incisor-canine-
premolar region, of which the canine region is the
most common site.3-5,8 The lesion usually appears
Introduction radiographically as a unilocular lesion but at least
‘The adenomatoid odontogenic tumour is a rare four cases of multilocular appearance have been
tumour that comprises only 0.1 per cent of tumours reported. 2,4,8 Radiopacities are often seen in the peri-
and cysts of the jaw and 3 per cent of all odontogenic coronal radiolucency. This phenomenon occurred in
tumours’.1 A most comprehensive review of the 65 per cent of the cases Giansanti et al. 2 reviewed in
odontogenic adenomatoid tumour was carried out which radiographs were available, or where mention
in 1970 by Giansanti and colleagues2 who sur veyed of the radiographic appearance was made. In one
three cases. In 1975, Courtney and Kerr3 reported case, irregular root resorption,11 and in two cases
20 additional cases. In 1981 Stroncek et al.4 exam- dilaceration, were reported.10,14 The size of the lesion
ined 37 cases reported in the English literature. In usually varied from 15 to 30 mm in diameter.
addition, in 1990, Toida et al.5 reviewed 126 Several larger tumours have been noted, the largest
Japanese cases. In all, 294 cases in the literature were was more than 120 mm.10 Radiographically, the
reviewed. The lesion is most frequently encountered lesion frequently looked like a dentigerous cyst or
in the second decade of life,1-20 with 19 years being follicular cyst.1-20 The radiolucency associated with
the mean of the cases reviewed. A range of ages from the odontogenic adenomatoid tumour may extend
36 to 82 years has been reported in the literature. more apically than the dentigerous cyst.3 An intra-
oral or extraoral swelling was the main symptom,
*Assistant Professor, Department of Oral and Maxillofacial Surgery,
and the swelling was usually painless and slow
Faculty of Dentistry, Atat¨ rk University, Turkey.
†Researcher, Department of Oral and Maxillofacial Surgery, Faculty
of Dentistry, Atat¨ rk University, Turkey.
‡Associate Professor and Chairman, Department of Oral Diagnosis Case report
and Radiology, Faculty of Dentistry, Atat¨ rk University, Turkey.
§Assistant Professor, Department of Pathology, School of Medicine,
A 15 year old girl presented with a swelling in the
Atat¨ rk University, Turkey.
u lateral-canine area of upper right jaw. She had visited
Australian Dental Journal 1997;42:5. 315
Fig. 1.–Intraoral radiograph showing irregular root resorption in borders of the lesion and in first premolar.
Small radiopaque calcifications can be seen in the lesion.
a dentist a year ago. The dentist without having a In the root of the lateral incisor in the lesion,
radiograph had started the treatment by extracting dilaceration was seen (Fig. 2). There was irregular
her upper deciduous lateral incisor and cuspid teeth root resorption in the right maxillary first premolar.
and by applying antibiotic medication. On realizing The lesion was directly associated with both the nose
that the swelling was getting worse, the patient and sinus. Aspiration was attempted and yielded 5
presented at the Oral Diagnosis and Radiology mL of a turbid grey-tan fluid. Under local
Clinic of the Dentistry Faculty, Atat¨ rk University in anaesthesia the lesion and impacted lateral incisor
April 1993. and canine were extracted.
Extraoral examination disclosed a swelling in the After the operation the specimen was fixed in 10
anterior maxilla with no pain. When examined intra- per cent formal saline and prepared for histological
orally it was seen that the right maxillary lateral examination. Some sections were stained with
incisor and canine teeth were missing and there was haematoxylin-eosin, while others were stained with
a hyperaemic swelling in the vestibule. Congo red and crystal violet.
On radiological examination (intraoral panoramic Microscopically a poor connective tissue stroma
radiography, Waters sinus occlusal periapical views) was seen. Spindle or polyhedral epithelial cells in
a radiolucent lesion with a regular border was seen this stroma displayed duct-like structures. In some
in the area ranging between the right maxillary areas amorphous eosinophilic material was seen
central and right first molar. The lesion was among the tumour cells in the form of solid nests.
45 40 mm in size and was unilocular and showed This material showed a positive reaction with the
small radiopaque calcification points. In the lesion, Congo red dye, but with the crystal violet the reaction
the maxillary canine was impacted in the apex of was negative. Microcalcifications were seen in all
first molar tooth and lateral incisor (Fig. 1). regions of the tumour (Figs. 3-6).
It was Stafne who identified the adenomatoid
odontogenic tumour for the first time in 1948.2,4,9,10
Subsequently, in cases reported by various authors4-6
these tumours are described as intraoral-extraoral
swellings in the maxilla2,5,9,10 which generally are
more frequently seen in females2-5 and which mostly
occur in the second decade of life.1-20 Apart from a
few exceptional cases4,5 the tumour is associated with
unerupted teeth. The unerupted teeth are usually
canine or lateral incisors.2-5,8 Irregular root resorption
and dilaceration within the lesion are only infrequently
reported in the literature.10,14 Clinical, radiographic
5 mm and macroscopic findings in the present case are
Fig. 2.–Dilaceration in root of lateral incisor in tumour, and canine
consistent with descriptions of the lesion in the
displaced in tumour. Bar=5 mm. dental literature. It was also observed that the
316 Australian Dental Journal 1997;42:5.
Fig. 3.–Calcification and duct-like structures. H&E. 40.
Fig. 4.–Duct-like and adenoid structures. H&E. 100.
Fig. 5.–Tumour stroma and duct-like structures. H&E. 100.
Fig. 6.–Amyloid-like mid-substance. H&E. 100.
present tumour was associated with two unerupted 04. Stroncek GG, Acevedo A, Higa LH. An atypical odontogenic
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05. Toida M, Hytodo I, Okuda T, et al. Adenomatoid odontogenic
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