CASE REPORT

A case of glandular odontogenic cyst associated with

ameloblastoma: correlation of diagnostic imaging with
histopathological features
M Hisatomi*
,1
, J Asaumi
1
, H Konouchi
1
, Y Yanagi
1
and K Kishi
1
1
Department of Oral Radiology, Okayama University Dental School, Okayama, Japan
The glandular odontogenic cyst (GOC) is a rare odontogenic cyst. There has only been one
reported case of the simultaneous presence of GOC and ameloblastoma. The radiographic
features of GOC are well established but the MR ndings have not been described. We report a
case of GOC associated with ameloblastoma with special reference to the correlation of the
diagnostic imaging with the histopathological features.
Keywords: magnetic resonance imaging; jaw cysts; jaw neoplasms; jaw diseases
Case report
A 45-year-old woman was referred by her dentist for
evaluation of pain on biting and mobility of the right
mandibular premolars that had lasted approximately a
year. No relevant medical history was recorded. The
gingiva was normal in appearance but there was
paresthesia of the right lower lip.
Imaging features
Panoramic and periapical radiographs showed a 3 cm
diameter, well-dened, unilocular radiolucency below
the right canine and premolars (Figure 1a). The apices
of all three teeth were resorbed. A true occlusal
radiograph and axial CT scan showed loss of
continuity of the buccal cortex with thinning and
slight expansion lingually (Figure 1b).
MR examination was performed on a 1.5 tesla unit
(Magnetom Vision, Siemens, Erlangen, Germany) with
a CP head coil. T1 and T2-weighted spin-echo
sequences (T1WI, TR/TE=600/15 ms, and T2WI,
TR/TE=3000/105 ms) were obtained before adminis-
tration of contrast. Slice thickness was 4 mm and the
matrix 1966256 pixels. Muscle signal was interpreted
as intermediate on T1WI, and cerebrospinal uid as
high signal intensity on T2WI.
Gadopentetate dimeglumine (Gd-DTPA) (Magne-
vist, Nihon Schering, Osaka, Japan) was injected as a
bolus at approximately 2 ml/s. The beginning of rst
scan was designated as time 0 and administration of
Gd-DTPA was started 10 s before the second scan.
Dynamic Gd-T1WI images were acquired as 11
consecutive scans at 1 s intervals (29 s/1 scan). The
signal intensity of the lesion was measured on the
images from each scan and the time-signal intensity
curve (TIC) plotted.
In addition, T1WI images were obtained after
intravenous injection of Gd-DTPA (Gd-T1WI) in the
same way as before its administration.
The lesion was divided into three portions on the
basis of the signal intensity (Figure 2a c). The mesial
part (area A) had intermediate signal intensity on
T1WI, high on T2WI, and a rim-enhancement on Gd-
T1WI. The buccal portion of the distal part (area B)
showed low signal intensity on T1WI, intermediate on
T2WI and homogeneous enhancement on Gd-T1WI.
The lingual portion of the distal part (area C) showed
high signal intensity on both T1WI and T2WI and
homogeneous enhancement on Gd-T1WI.
The TIC curve of area A could not be assessed
because the enhanced area was so small that it was not
possible to set up a region of interest. TIC of area B
*Correspondence to: M Hisatomi, Okayama University Dental School, 2-5-1,
Shikata-Cho, Okayama City, Okayama, 700 8525, Japan
Received 26 January 2000; accepted 21 March 2000
Dentomaxillofacial Radiology (2000) 29, 249 253
2000 Macmillan Publishers Ltd. All rights reserved 0250 832X/00 $15.00
www.nature.com/dmfr
a b
Figure 1 (a) Periapical radiograph showing an unilocular radiolucency with an irregular outline associated with resorption of the apices of the
right mandibular canine and premolars. (b) CT scan (bone window) showing expansion and loss of continuity of the buccal cortex of the
mandible. The lingular cortex is thinned and slightly expanded
a b c
Figure 2 Axial MR scans. (a) T1WI, (b) T2WI, (c) Gd-T1WI. Area A showed intermediate signal intensity on T1WI, high on T2WI and rim-
enhancement on Gd-T1WI. Area B showed low signal intensity on T1WI, intermediate on T2WI and enhancement on Gd-T1WI. Area C
showed high signal intensity on both T1WI and T2WI and enhancement on Gd-T1WI
GOC with ameloblastoma
M Hisatomi et al
250
Dentomaxillofacial Radiology
rapidly increased to reach a plateau and then declined
slowly. The TIC of area C gradually increased and
then gradually decreased (Figure 3).
Histopathological ndings
Histopathologically, the lesion consisted of a cystic
portion and a solid portion. The solid portion
consisted of dispersed islands of odontogenic
epithelium with an abundant stroma containing
dense collagen bundles (Figure 4a,b). The islands
varied in shape and size and the cells resembled the
enamel organ. The epithelial islands were arranged
as cuboidal or columnar ameloblast-like cells and the
centre of the epithelial islands were composed of
cells resembling stellate reticulum. These appearances
were considered to be those of a typical amelo-
blastoma with mixed follicular and plexiform
patterns.
The cystic portion was lined by stratied epithelium
with irregular papillary extrusions on the inner surface
(Figure 4c). The inner layer consisted of columnar
cells and included cilia (Figure 4d). Several weakly
stained mucous cells and eosinophilic cuboidal cells
were visible within the epithelial layer (Figure 4e). The
basal cells of the epithelial layer were hyperchromatic,
suggesting odontogenic epithelium. The diagnosis was
GOC.
Discussion
The glandular odontogenic cyst (GOC) is a rare
odontogenic cyst which was rst reported by
Gardner et al. in 1988.
1
Histopathologically, some
authors have reported that the lesion contains
glandular structures within the basal and/or the
spinous cell layers,
1,2
though in our case this region
did not appear to be vacuolated. The presence of
mucous cells and cilia were consistent with GOC.
The solid portion in our case, showed the
characteristic features of mixed plexiform and
follicular ameloblastoma.
Gardner et al. reported a case of GOC associated
with ameloblastoma,
1
with ndings similar to ours.
Raubenheimer et al. described a follicular ameloblas-
toma which showed acanthomatous dierentiation and
foci of mucous cell metaplasia.
3
Although the tumor
cells in ameloblastoma have a high ability to undergo
varying forms of metaplastia,
4
it is unusual to nd
mucous cells. In terms of these histopathological
ndings, our case was not similar to that of
Raubenheimer et al.
3
There is a possibility that the
tumor cells in the ameloblastoma can metamorphose
into mucous cells in response to inammatory stimuli
although no inammatory inltrate was detected
within the brous connective tissue, and our lesion
had cilia which are not seen in ordinary ameloblas-
toma. Therefore, in our case, the nal diagnosis of
GOC associated with ameloblastoma was established
by the presence of mucous cells and cilia within the
epithelia.
Plain radiographs in our case showed a well-dened
unilocular radiolucent lesion, similar in appearance to
the case of GOC associated with ameloblastoma
described by Gardner et al.
1
However, it was dicult
to dierentiate the areas of GOC and ameloblastoma
using CT or plain views. On MR, area A, the cystic
portion corresponded to GOC since the signal
intensities were similar to those of other odontogenic
cysts.
5,6
The lining epithelium was enhanced, possibly
due to rich microvessels within the epithelia. However,
because it was so small, dynamic MR was non-
contributory.
Histologically, area B, the buccal region of the solid
portion, consisted of odontogenic epithelial islands of
various shapes and abundant collagenized stroma. The
signal intensity on T1WI and T2WI was thought to
reect the characteristics of the stroma. The depiction
of the epithelial islands in this part of the lesion was
enhanced on Gd-T1WI.
Unfortunately, the condition of the specimen in area
C, the lingual region of the solid portion, was poor.
Therefore, we could not correlate the histopathogical
ndings with its signal intensity.
Dynamic MRI is a new method for the assessment
of organ perfusion.
7
In most benign tumors of the
maxillofacial region, TIC shows a slow increase.
8
Area
B, however, showed a rapid increase and delayed
washout pattern, reecting the histopathological
features of rich tumor cells and abundant micro-
vessels. We could not elucidate why the signal
intensities on MR images and TIC diered between
areas B and C due to the poor condition of the
specimen from area C.
In conclusion, we report a case of GOC associated
with ameloblastoma and correlate the diagnostic
imaging, especially MRI, with its histopathological
features. MRI was useful to dierentiate the cystic
from the solid portion of the lesion. The MR features
Figure 3 Time-signal intensity curves (TIC) obtained by dynamic
MRI for areas B and C. TIC of area B rapidly increased to reach a
plateau and then decreased whereas area C gradually increased and
then gradually decreased. ^ area B, & area C
Dentomaxillofacial Radiology
GOC with ameloblastoma
M Hisatomi et al
251
of the ameloblastoma corresponded with the histo-
pathological ndings. However, it was not possible to
demonstrate the characteristic features of the cystic
lesion of GOC on MRI. Further study of GOC is
necessary to improve MR sequences and obtain more
detailed information.
a
c
b
d
e
Figure 4 Histopathological ndings. (a) Photomicrograph of the solid portion showing the epithelial islands with an abundant stroma
containing dense collagen bundles (H&E stain magnication625). (b) Higher powered view showing the follicular pattern in the epithelial
islands (H&E stain magnication6250). (c) Photomicrograph of the cystic portion showing the stratied epithelium with an irregular papillary
extrusion (H&E: magnication6250). (d) Higher powered view showing eosinophilic cuboidal cells and presence of cilia within the epithelium
(H&E: magnication6500). (e) Higher powered view showing several weakly staining mucous cells within the epithelium (mucicar-
mine : magnication6500)
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GOC with ameloblastoma
M Hisatomi et al
252
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