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Australian Dental Journal 1997;42:(5):315-8

Adenomatoid odontogenic tumour


(adenoameloblastoma). Case report and review of the
literature
Ertun Day, DDS, PhD*
Gelengul Gurbuz, DDS
O. Murat Bilge, DDS, PhD
M. Akif iftcioglu, MD, PhD

Abstract
An adenomatoid tumour was found in the anterior
maxillary region of a 15 year old female patient.
Two impacted teeth were found in the tumour. The
lateral incisor found in the tumour was dilacerated,
and the roots of the first premolar were resorbed. A
review of the English literature indicated that 294
similar cases have been reported.
Key words: Adenomatoid odontogenic tumour, case
report.
(Received for publication March 1994.
December 1994. Accepted January 1995.)

Revised

Introduction
The adenomatoid odontogenic tumour is a rare
tumour that comprises only 0.1 per cent of tumours
and cysts of the jaw and 3 per cent of all odontogenic
tumours.1 A most comprehensive review of the
odontogenic adenomatoid tumour was carried out
in 1970 by Giansanti and colleagues2 who sur veyed
three cases. In 1975, Courtney and Kerr3 reported
20 additional cases. In 1981 Stroncek et al.4 examined 37 cases reported in the English literature. In
addition, in 1990, Toida et al.5 reviewed 126
Japanese cases. In all, 294 cases in the literature were
reviewed. The lesion is most frequently encountered
in the second decade of life,1-20 with 19 years being
the mean of the cases reviewed. A range of ages from
36 to 82 years has been reported in the literature.
*Assistant Professor, Department of Oral and Maxillofacial Surgery,
Faculty of Dentistry, Ataturk University, Turkey.
Researcher, Department of Oral and Maxillofacial Surgery, Faculty
of Dentistry, Atatu rk University, Turkey.
Associate Professor and Chairman, Department of Oral Diagnosis
and Radiology, Faculty of Dentistry, Ataturk University, Turkey.
Assistant Professor, Department of Pathology, School of Medicine,
Ataturk University, Turkey.
Australian Dental Journal 1997;42:5.

The tumour affects females more than males in


almost a two to one ratio.2-5 The maxilla is involved
nearly twice as frequently as the mandible.2-5,9,10
Unerupted permanent teeth were associated with
this lesion in one-third of the cases.2-5 In a few cases,
more than one unerupted tooth was associated with
the tumour.4,5
The cuspid is the tooth most commonly associated
with the adenomatoid odontogenic tumour.4,5,8,11
Three-quarters of the tumours involved the anterior
aspect of the jaws, particularly the incisor-caninepremolar region, of which the canine region is the
most common site.3-5,8 The lesion usually appears
radiographically as a unilocular lesion but at least
four cases of multilocular appearance have been
reported. 2,4,8 Radiopacities are often seen in the pericoronal radiolucency. This phenomenon occurred in
65 per cent of the cases Giansanti et al. 2 reviewed in
which radiographs were available, or where mention
of the radiographic appearance was made. In one
case, irregular root resorption,11 and in two cases
dilaceration, were reported.10,14 The size of the lesion
usually varied from 15 to 30 mm in diameter.
Several larger tumours have been noted, the largest
was more than 120 mm.10 Radiographically, the
lesion frequently looked like a dentigerous cyst or
follicular cyst.1-20 The radiolucency associated with
the odontogenic adenomatoid tumour may extend
more apically than the dentigerous cyst.3 An intraoral or extraoral swelling was the main symptom,
and the swelling was usually painless and slow
growing.5,7,13
Case report
A 15 year old girl presented with a swelling in the
lateral-canine area of upper right jaw. She had visited
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


radiograph had started the treatment by extracting
her upper deciduous lateral incisor and cuspid teeth
and by applying antibiotic medication. On realizing
that the swelling was getting worse, the patient
presented at the Oral Diagnosis and Radiology
Clinic of the Dentistry Faculty, Ataturk University in
April 1993.
Extraoral examination disclosed a swelling in the
anterior maxilla with no pain. When examined intraorally it was seen that the right maxillary lateral
incisor and canine teeth were missing and there was
a hyperaemic swelling in the vestibule.
On radiological examination (intraoral panoramic
radiography, Waters sinus occlusal periapical views)
a radiolucent lesion with a regular border was seen
in the area ranging between the right maxillary
central and right first molar. The lesion was
45 40 mm in size and was unilocular and showed
small radiopaque calcification points. In the lesion,
the maxillary canine was impacted in the apex of
first molar tooth and lateral incisor (Fig. 1).

5 mm

Fig. 2.Dilaceration in root of lateral incisor in tumour, and canine


displaced in tumour. Bar=5 mm.
316

In the root of the lateral incisor in the lesion,


dilaceration was seen (Fig. 2). There was irregular
root resorption in the right maxillary first premolar.
The lesion was directly associated with both the nose
and sinus. Aspiration was attempted and yielded 5
mL of a turbid grey-tan fluid. Under local
anaesthesia the lesion and impacted lateral incisor
and canine were extracted.
After the operation the specimen was fixed in 10
per cent formal saline and prepared for histological
examination. Some sections were stained with
haematoxylin-eosin, while others were stained with
Congo red and crystal violet.
Microscopically a poor connective tissue stroma
was seen. Spindle or polyhedral epithelial cells in
this stroma displayed duct-like structures. In some
areas amorphous eosinophilic material was seen
among the tumour cells in the form of solid nests.
This material showed a positive reaction with the
Congo red dye, but with the crystal violet the reaction
was negative. Microcalcifications were seen in all
regions of the tumour (Figs. 3-6).
Discussion
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
and macroscopic findings in the present case are
consistent with descriptions of the lesion in the
dental literature. It was also observed that the
Australian Dental Journal 1997;42:5.

6
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


teeth and that there was resorption in the first
premolar and dilaceration of the lateral incisor.
According to Giansanti et al.2 after local curettage of
the tumour a number of cases were followed up for
periods ranging from one to ten years with no
reported cases of recurrence. Indeed, Giansanti et
al.2 reported that the adenomatoid odontogenic
tumour was a completely benign tumour which
never recurred once removed. In the one year
follow-up in the present case no recurrence was
reported.
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Address for correspondence/reprints:


Dr O. M. Bilge,
Department of Oral Diagnosis and Radiology,
Faculty of Dentistr y,
Ataturk University,
Erzurum,
Turkey.

ustralian Dental Journal 1997;42:5.