Open Journal of Stomatology, 2013, 3, 504-506 OJST
http://dx.doi.org/10.4236/ojst.2013.39082 Published Online December 2013 (http://www.scirp.org/journal/ojst/)
Calcifying cystic odontogenic tumour manifesting in the
maxillary antrum: Case report
Eunice Kihara1, Richard Owino2, Josiah Otwoma1, Elizabeth Dimba1, Mark Chindia1
1Department of Oral & Maxillofacial Surgery—Oral Pathology & Oral Medicine, University of Nairobi, Nairobi, Kenya
2Department of Paediatric Dentistry & Orthodontics, University of Nairobi, Nairobi, Kenya
Received 31 October 2013; revised 1 December 2013; accepted 12 December 2013
Copyright © 2013 Eunice Kihara et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
A case is presented of a 15-year-old boy who mani-
fested the calcifying cystic odontogenic tumour (CCOT)
in the left maxillary antrum of an unknown duration.
In addition, the patient had a high arched palate and
multiple impacted teeth of the normal series and su-
pernumerary type including mesiodens as demon-
strated in an orthopantomograph.
Keywords: Case Reports; Odontogenic Cyst; Calcifying;
Odontogenic Tumours; Maxillary Sinus
1. INTRODUCTION
Generally, benign odontogenic neoplasms are relatively
rare lesions that are derived from either the epithelial an d
mesenchymal components or the remnant elements of the
developing tooth germs. These lesions are often classi-
fied according to location as p eripheral or central entities
[1-4]. In this category of diseases, the calcifying cystic
odontogenic tumour (CCOT) is a particularly rare benign
entity that is characterized by an ameloblastoma-like
epithelium and ghost cells that have the potential to un-
dergo calcification [4-6]. The lesion usually appears as a
painless, slow-growing tumour that may involve the
maxilla or mandible in the anterior segments [1,3]. We
report a 15-year-old boy who presented with CCOT man-
ifesting in the left maxillary antrum associated with den-
tal malformation, tooth impaction and a high arched pal-
ate.
2. CASE REPORT
A 15-year-old boy was referred to our clinic for evalua-
tion and management of a persistent swelling of the left
maxilla that had, apparently, been noted nearly two years
previously. Over that period, the swelling had been es-
sentially painless. However, the patient reported that the
swelling had then become painful. In addition, he also
experienced a feeling of stuffiness in the left nostril. The
patient’s general medical and dental history was unre-
markable. On examination, the patient manifested a non-
tender mass causing facial asymmetry with fullness in
the left nasolabial fo ld extending towards the bod y of the
zygoma. Intraorally, there was total left maxillary sulcu-
lar fullness, a mixed dentition and a generalized malposi-
tioning of some teeth in both arches in addition to a high
arched palate. A panoramic radiograph rev ealed multiple
bony impacted teeth of the normal series and thos e of the
supernumerary type and mesiodens “Figure 1”. The ra-
diograph also demonstrated a remarkably expanded op-
acified left maxillary sinus.
Under a local anaesthetic, aspiration of the lesion
yielded a shimmering amber-coloured cystic fluid. Then
an open incisional biopsy was obtained which on histo-
pathological examination demonstrated a fibrous capsule
associated with odontogenic cells with low mitotic indi-
ces and no pleomorphism. In addition, there were armo-
phous areas of eosinophilic material interspersed within
the cells. Ghost cells were prominent “Figure 2”. The tu-
mour was, thereafter, totally extirpated under general
anaesthesia “Figure 3”. The patient remains well after
about a 1-year regular follow up.
3. DISCUSSION
The most recent WHO classification proposes the diag-
nostic designations CCOT and dentinogenic ghost cell
tumour (DGCT) to differentiate between the cystic and
solid lesion [5]. Remarkably, these lesions may occur in
association with other odontogenic tumours including, in
most instances, the adenomatoid odontogenic tumour [7],
ameloblastoma and odontoma [6] and odontogenic kera-
tocystic tumour [8]. Practically, then, obtaining an ade-
quate biopsy sp ecimen that is represen tative of the entire
lesion during histopathological analysis has to be man-

E. Kihara et al. / Open Journal of Stomatology 3 (2013) 504-506 505
Figure 1. Panoramic view demonstrating an opacified left max-
illary antrum, multiple impacted and supernumeraries as well
as a calcified mass apical to 23.
(a) (b)
Figure 2. (a) Thin stratified squamous epithelial lining overly-
ing a fibrous connective tissue stroma; (b) Deposits of eosino-
philic material resembling dentin, ghost cell masses without
nuclei and areas of calcification (H and E sections 100×).
Figure 3. Excised mass showing the thick capsule and white
calcific material.
datory. This is because should CCOT occur in associa-
tion with ameloblastoma, the management modality must
conform to that of th e letter so that und esirable morbid ity
is avoided.
As for the present case, the entire lesion was histo-
logically reexamined after it was extirpated. Intraopera-
tively the main challenge entailed clinically differentiat-
ing tumour lining from the normal paranasal lining tis-
sues in some areas. Fortunately, as has been described in
the literature [1,4] the cystic linin g of the tumour tends to
be uniformly thick and, therefore, easy to separate from
the normal tissues.
With regard to the demographic pattern of CCOT, it
has been reported that the lesion may manifest between
the ages of 5 and 75 years with a mean of 25 years [9]. In
a 30-year series of 60 CCOT cases [2], a male prepon-
derance was demonstrated (37 male and 23 female).
Overall, other than tooth impaction, a few other clinical
features have been described such as those in the present
case which may imply a syndromic pattern of the tumour
on rare occasions.
4. CONCLUSION
A 15-year-old male presented with a rare case of a max-
illary sinus CCOT which was associated with multiple
impacted and supernumerary teeth and a high arched
palate. Radiographic and histological examinations were
consistent with features of CCOT. The patient remains
well after the enucleation of the tumour. Although rare,
CCOT is an important clinical entity since it may con-
comitantly occur with other destructive lesions including
ameloblastoma and the keratocystic odontogenic tumour.
Clinicians should, therefore, be encouraged to present
these cases as they occur.
5. ACKNOWLEDGEMENTS
We are most grateful to Pamela Muhonja Kegode for her participation
in the management of the patient and Sally Musinde for preparing the
manuscript.
REFERENCES
[1] Johnson III, A., Fletcher, M., Gold, L. and Chen, S.-Y.
(1997) Calcifying odontogenic cyst: A clinicopathologic
study of 57 cases with Immunohistochemical Evaluation
for Cytokeratin. Journal of Oral and Maxillofacial Sur-
gery, 55, 679-683.
http://dx.doi.org/10.1016/S0278-2391(97)90574-7
[2] Habibi, A., Saghravanian, N., Salehinejad, J. and Jafar-
zadeh, H. (2011) thirty years clinicopathological study of
60 Calcifying cystic odontogenic tumours in Iranian
Population. The Journal of Contemporary Dental Prac-
tice, 12, 171-173.
[3] Utumi, E.R., Pedron, I.G., Silva, L.P., Machado, G.G.
and Rocha, A.C. (2012) Different manifestations of cal-
cifying cystic Odontogenic tumour. Einstein (Sao Paulo),
10, 366-370.
http://dx.doi.org/10.1590/S1679-45082012000300019
[4] De Lima, A.P., Kitakawa, D., Almeida, J.D., Brandao,
A.A.H. and Anbinder, A.L. (2012) Peripheral calcifying
odontogenic tumour of the maxillary gingiva. BMC Re-
search Notes, 5, 455-457.
http://dx.doi.org/10.1186/1756-0500-5-455
[5] Barnes, L., Everson, J.W., Reichert, P.A., et al.; World

E. Kihara et al. / Open Journal of Stomatology 3 (2013) 504-506 506
Health Organisation Classification of Tumours (2005)
Pathology and genetics of tumours of the head and neck.
Chapter 6. Odontogenic tumours. IARC, Lyon, 283-327.
[6] Slootweg, P.J. (2007) Dental pathology: A practical in-
troduction. Springer, Berlin.
[7] Zeitoun, I.M., Dhanrajani, P.J. and Masodomi, H.A.
(1996) Adenomatoid odontogenic tumour arising in cal-
cifying Odontogenic cyst. Journal of Oral and Maxillo-
facial Surgery, 54, 634-637.
http://dx.doi.org/10.1016/S0278-2391(96)90650-3
[8] Basile, J.R., Klene, C. and Lin, Y.L. (2010) Calcifying
odontogenic cyst with odontogenic keratocyst: A case
report and review of the literature. Oral Surgery, Oral
Medicine, Oral Pathology, Oral Radiology and Endodon-
tology, 109, e 4 0 -e45.
http://dx.doi.org/10.1016/j.tripleo.2009.12.026
[9] Daniels, J.S.M. (2006) Recurrent calcifying odontogenic
cyst involving the maxillary sinus. Oral Surgery, Oral
Medicine, Oral Pathology, Oral Radiology and Endodon-
tology, 101, 356-362.