Beruflich Dokumente
Kultur Dokumente
Case Report
a r t i c l e i n f o a b s t r a c t
Article history: We report a case of calcifying cystic odontogenic tumor (CCOT) with odontoma developing in the decid-
Received 31 August 2015 uous dentition period, and present previously reported cases in Japan. A 5-year-old boy visited a dental
Received in revised form 14 January 2016 clinic because of swelling on his left mandibular buccal alveolar region, and was referred to our depart-
Accepted 31 March 2016
ment. Clinical examination revealed a 20-mm (in diameter), bone-like, hard mass on the left mandibular
Available online 4 May 2016
buccal gingiva at the rst deciduous molar region. A panoramic radiograph showed a 25 mm 20 mm,
unilocular, cystic radiolucent area from the left mandibular second deciduous molar to the rst molar
Keywords:
region. There were radiopaque tiny ecks at the upper side of the lesion. The clinical diagnosis was a
Calcifying cystic odontogenic tumor
Odontoma
benign tumor of the left mandible. Consequently, tumor enucleation was performed under general anes-
Deciduous dentition thesia. Histopathological diagnosis conrmed a CCOT associated with odontoma. Five years after surgery,
the left mandibular region has healed well, with no evidence of recurrence.
2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.
1. Introduction the deciduous dentition period in a 5-year-old boy, and discuss the
relevant literature.
Calcifying cystic odontogenic tumor (CCOT) is a disease display-
ing neoplastic potential and is characterized by calcication of the 2. Case report
epithelium and the presence of ghost cells. Although various sus-
ceptible ages have been reported, CCOT tends to occur frequently The patient is a 5-year-old boy. In November 2008, he visited a
in the young. However, cases of CCOT in patients aged <10 years dental clinic to undergo radiography for dental caries. These images
are rare; in particular, there is only one reported case in Japan of indicated the presence of a radiolucent area in the left mandible,
CCOT occurring in the deciduous dentition period [1]. We report and the patient was referred to our department for detailed exami-
our experience of a case of CCOT with odontoma developing in nation. He did not present with any particular past history or family
history of note. He had a height of 100 cm, weight of 15 kg, and
low body mass index, but good nutritional status. The external
orofacial appearance showed bilateral asymmetry. Regional lymph
nodes were without swelling or tenderness. The left mental nerve
Abbreviation: CCOT, calcifying cystic odontogenic tumor.
Asian AOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian region did not demonstrate hypoesthesia. The internal oral cavity
Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathol- examination revealed a bone-like, hard mass 20 mm in diameter in
ogy; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese the buccal alveolar region of the left mandibular second deciduous
Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants. molar; however, no parchment-like appearance was observed.
Corresponding author. Tel.: +81 86 235 6697; fax: +81 86 235 6699.
A panoramic radiograph showed a 25 mm 20 mm, unilocular,
E-mail addresses: hachi70@md.okayama-u.ac.jp (N. Moritani),
n-nakata@md.okayama-u.ac.jp (N. Nakata), eikiyama@md.okayama-u.ac.jp
cystic radiolucent area from the left mandibular second decid-
(E. Yamachika), tatsushi@md.okayama-u.ac.jp (T. Matsumura), uous molar periapical region to the rst molar region. There
jin@md.okayama-u.ac.jp (H. Nagatsuka), iida-s1@cc.okayama-u.ac.jp (S. Iida). were radiopaque tiny ecks at the upper side of the area. The
http://dx.doi.org/10.1016/j.ajoms.2016.03.009
2212-5558/ 2016 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.
422 N. Moritani et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 421425
3. Discussion
Case Age/sex Region Appearance With or Impacted tooth Relationship Deviation of Root resorption of Diagnosis Treatment Treatment of Prognosis Author (y)
without inside the between lesion impacted tooth approximal tooth impacted tooth (follow-up
odontoma lesion and impacted caused by lesion in contact with associated period)
N. Moritani et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 421425
tooth lesion with lesion
1 9/F Right maxilla Unilocular With Supernumerary Inside lesion Not mentioned Not mentioned Dentigerous Extirpation Extraction of Not Eda et al.
tooth-like cyst supernumer- recorded (1974) [15]
deciduous ary
molar tooth
2 9/M Left mandible Unilocular With Left Inside lesion None None Dentigerous Extirpation Extraction of Good (3 y 2 Igari et al.
mandibular cyst left m) (1982) [16]
canine mandibular
canine
3 9/F Left maxilla Unilocular With Left maxillary Outside lesion Left maxillary None Maxillary cyst Extirpation Extraction of Good (1 y 8 Sato et al.
second molar rst molar after left maxillary m) (1982) [17]
fenestration rst and
second molar
4 9/F Right Unilocular With None Outside lesion Left mandibular Left mandibular Maxillary cyst Extirpation Extraction of Good (6 y 3 Misaki
mandible-left lateral deciduous lateral or odontogenic left m) et al.
mandible incisor-second incisor, deciduous tumor mandibular (1986) [9]
premolar, right second molar lateral incisor
mandibular
canine-rst
premolar
5 7/F Right Unilocular Without Right Inside lesion Right mandibular Not mentioned Dentigerous Extirpation Extraction of Good (1 y Fujisawa
mandible mandibular canine, second cyst after right 11 m) et al.
rst premolar premolar fenestration mandibular (1991) [7]
rst premolar
6 4/F Right maxilla Unilocular With Deciduous Outside lesion None None Compound Extirpation Conservation Good (7 m) Murakami
maxillary right odontoma et al.
incisor (1991) [1]
7 7/M Right maxilla Unilocular With None Outside lesion Right maxillary None Maxillary cyst Extirpation Conservation Good (1 y 9 Eda et al.
canine, second after m) (2003) [18]
molar fenestration
8 6/M Right maxilla Unilocular With None Outside lesion Right mandibular None Compound Extirpation Conservation Good (9 m) Hayashi
canine, rst odontoma or et al.
premolar dentigerous (2004) [6]
cyst with
compound
odontoma
9 5/M Left mandible Unilocular With None Outside lesion Left mandibular Left mandibular Left Extirpation Conservation Good (5 y) Present
second premolar, second molar mandibular case
rst molar tumor
N. Moritani et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 421425 425
teeth in CCOT is considered appropriate. On the other hand, there [2] Gorlin RJ, Pindborg JJ, Clausen FP, Vickers RA. The calcifying odontogenic cyst
were ve cases of CCOT in children aged <10 years with deviated a possible analogue of the cutaneous calcifying epithelioma of Malherbe. Oral
Surg Oral Med Oral Pathol 1962;15:123543.
impacted teeth adjacent to the CCOT. In one of these, Sato et al. [17] [3] Freedman PD, Lumerman H, Gee JK. Calcifying odontogenic cyst. A review and
extracted the deviated impacted teeth; in another, Misaki et al. [9] analysis of seventy cases. Oral Surg Oral Med Oral Pathol 1975;40:93105.
reported that one of ve deviated impacted teeth was extracted. [4] Pratorious F, Ledesma-Montes C. Calcifying cystic odontogenic tumour. In:
Barnes L, Eveson JW, Reichart PS, Idransky D, editors. World Health Orga-
In cases in which impacted teeth were preserved [1,6,9,18], the nization Classication of Tumours. Pathology and genetics of head and neck
teeth were expected to erupt; however, these reports say little tumours. Lyon: IARC Press; 2005. p. 22740.
about prognosis. In a case reported by Eda et al. [18], the devi- [5] Ledesma-Montes C, Gorlin RJ, Shear M, Prae Torius F, Mosqueda-Taylor A,
Altini M, et al. International collaborative study on ghost cell odontogenic
ated impacted teeth demonstrated a tendency to return to their
tumours: calcifying cystic odontogenic tumour, dentinogenic ghost cell tumour
normal positions, and root formation was observed. Misaki et al. and ghost cell odontogenic carcinoma. J Oral Pathol Med 2008;37:3028.
[9] also wrote that all of the preserved impacted teeth in their case [6] Hayashi Y, Katakura A, Kurosu M, Matsuzaka K, Yakushiji M. A case report of a
calcifying odontogenic cyst associated with odontoma in a child. Jpn J Ped Dent
erupted normally. In our own case as well, the deviated impacted
2004;42:44752.
teeth erupted at normal positions, and we observed no abnormal- [7] Fujisawa K, Nakanishi H, Satomura K, Yasuda K, Miyamoto Y, Kagayama M,
ities in root formation. Therefore, impacted teeth deviated by a et al. Three cases of calcifying odontogenic cyst. J Jpn Stomatol Soc 1991;40:
tumor can be expected to erupt, thus making preservation of these 90011.
[8] Poleti ML, Duarte BG, Lara VS, Freitas-Faria P, Rubira-Bullen IR, Goncales ES.
teeth appropriate. The only cases that demonstrated root resorp- Odontoma associated with calcifying cystic odontogenic tumor in deciduous
tion adjacent to the CCOT were a case reported by Misaki et al. dentition: case report. Oral Maxillofac Surg 2013;17:7780.
[9] and our own. A patient with a massive CCOT in the median [9] Misaki T, Miyake M, Hori M, Kudo I, Araki M, Sadanaga T, et al. Three cases of
calcifying odontogenic cyst. Jpn J Oral Maxillofac Surg 1986;32:57292.
mandible reported by Misaki et al. [9] demonstrated resorption of [10] Uchiyama Y, Akiyama H, Murakami S, Koseki T, Kishino M, Fukuda Y, et al.
roots of multiple erupted deciduous teeth; all of these teeth were Calcifying cystic odontogenic tumour: CT imaging. Br J Radiol 2012;85:54854.
ultimately extracted. Basically, extraction is considered appropri- [11] Abrams AM, Howell FV. The calcifying odontogenic cyst. Report of four cases.
Oral Surg Oral Med Oral Pathol 1968;25:594606.
ate for erupted teeth that demonstrate root resorption due to a [12] Freedman PD, Lumerman H, Gee JK. Calcifying odontogenic cyst. A review and
tumor. However, in the present case, the distal root of the left analysis of seventy cases. Oral Surg Oral Med Oral Pathol 1975;40:93106.
mandibular second deciduous molar (the tooth that demonstrated [13] Tanimoto K, Tomita S, Aoyama M, Furuki Y, Fujita M, Wada T. Radiographic
characteristics of the calcifying odontogenic cyst. Int J Oral Maxillofac Surg
root resorption) did not demonstrate adhesion to the lesion during
1988;17:2932.
surgery; the tumor was extirpated en bloc, and the possibility of [14] Iida S, Fukuda Y, Ueda T, Aikawa T, Arizpe JE, Okura M. Calcifying odontogenic
residual tumor cells was considered low. Also, because the teeth in cyst: radiologic ndings in 11 cases. Oral Surg Oral Med Oral Pathol Oral Radiol
Endod 2006;101:35662.
question are integral to occlusion in the deciduous dentition period,
[15] Eda S, Yanagisawa Y, Koike H, Yamamura T, Kato T. Two cases of calcifying
preserving them was considered advantageous for the growth and odontogenic cyst associated with odontoma, with an electron-microscopic
development of dentition and occlusion. Therefore, preserving the observation. Bull Tokyo Dent Coll 1974;15:7790.
teeth rather than extracting them led to extremely favorable post- [16] Igari T, Echigo S, Matsuda K, Abe Y, Teshima T. A report of two cases of calcify-
ing odontogenic cyst and a review of the literature. Jpn J Oral Maxillofac Surg
operative progress; thus, preservation was believed to have been 1982;28:123946.
valid in the present case. Although CCOT generally has a favorable [17] Sato O, Horikoshi M, Tagami Y, Tagami T, Sugiyama Y, Nagura H, et al. A case of
prognosis, there have been cases of postoperative recurrence [18], calcifying odontogenic cyst associated with tumorous proliferation. Jpn J Oral
Maxillofac Surg 1982;28:21019.
as well as cases of malignant transformation during recurrence [23]. [18] Eda A, Shigematsu H, Suzuki S, Kusama K, Sakashita H. A case of calcify-
However, in CCOT occurring during the deciduous dentition period, ing odontogenic cyst associated with odontoma in a 7-year-old boy. POMS
there have been no reported cases of recurrence or malignant trans- 2003;13:217.
[19] Altini M, Farman AG. The calcifying odontogenic cyst. Eight new cases and a
formation; thus, the prognosis is presumed to be favorable. On the review of the literature. Oral Surg Oral Med Oral Pathol 1975;40:7519.
other hand, there have been no reports of long-term follow-up; [20] Chindasombatjaroen J, Poomsawat S, Klongnoi B. Calcifying cystic odon-
therefore, the present case requires ongoing observation. togenic tumor associated with other lesions: case report with cone-beam
computed tomography ndings. Oral Surg Oral Med Oral Pathol Oral Radiol
2012;113:41420.
Conict of interest [21] Hamada Y, Miyazono K, Hashimoto T, Morita S, Inoue M, Fukutake Y, et al.
Three cases of calcifying odontogenic cyst, including the recurrent case. Jpn J
Oral Maxillofac Surg 1983;29:113344.
None of the authors have any conict of interest to declare.
[22] Chiba K, Kudo K, Ogawa K, Oguchi Y, Madarame Y, Fujioka Y, et al. Clinico-
pathological investigation of the cysts of the jaw of young patients with the
References special reference to the effects of marsupialization. Jpn J Oral Maxillofac Surg
1977;23:7717.
[23] Tarakji B, Ashok N, Alzoghaibi I, Altamimi MA, Azzeghaiby SN, Baroudi K, et al.
[1] Murakami A, Shintani S, Izumitani A, Ooshima T, Sobue S, Ishida T. A case of a
Malignant transformation of calcifying cystic odontogenic tumour a review
calcifying odontogenic cyst associated with odontoma inducing impaction of a
of literature. Contemp Oncol (Pozn) 2015;19:1846.
deciduous tooth. Jpn J Ped Dent 1991;29:1815.