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Case Report
Squamous Odontogenic Tumor: Diagnosis
and Management
Kamran Haghighat,* John R. Kalmar,† and Angelo J. Mariotti*‡

The squamous odontogenic tumor (SOT) is a rare, The squamous odontogenic tumor (SOT) is a rare
benign, locally infiltrative neoplasm of the jaws that pathologic entity that was originally defined and
appears to originate from the rests of Malassez. It has named in 1975 by Pullon et al.1 as a series of 6 pre-
been confused with other pathologic entities such as viously undescribed lesions. Since that time, fewer
ameloblastomas, carcinomas, and fibromas and clin- than 50 cases have been described in the literature.
ically may resemble localized periodontal disease. The The tumor typically arises as a solitary lesion in the
tumor is often asymptomatic, although it can present third decade of life, however, multifocal cases have
with symptoms of pain and tooth mobility. A char- also been described.1,2 Favored sites include maxil-
acteristic radiographic appearance is that of a trian- lary canine and mandibular premolar regions of the
gular-shaped or semi-circular lucency associated with jaw. The tumor exhibits indolent growth and is usu-
the roots of erupted teeth. Histologically, the tumor is ally asymptomatic. Occasionally local pain, gingival
characterized by the formation of variably sized nests swelling, and mobility of adjacent teeth have been
and cords of uniform, benign-appearing, squamous reported. The maxillary lesions seem to be more
epithelium with occasional vacuolization and kera- aggressive in nature than tumors located in the
tinization. Treatment of SOT by conservative surgical mandible. No gender or racial predilection of this neo-
excision is normally curative with rare episodes of plasm has been described.
recurrence reported. Since the clinical presentation of Radiographically, SOT often exhibits a character-
SOT may mimic more common pathologic entities, istic unilocular and triangular-shaped radiolucency of
this case report reinforces the need for careful histo- the alveolar bone, with the wide base of the lucency
logic evaluation of all lesions found in the periodon- localized between the diverging apices of the adjacent
tium. J Periodontol 2002;73:653-656. roots. Occasionally, scalloping and saucerization of
the underlying bone is seen; however, this is regarded
KEY WORDS
as a pressure phenomenon rather than tumor infil-
Neoplasms, squamous cell; odontogenic tration.3
tumors/diagnosis. In this case report, we document the clinical, radi-
ographic, and histologic characteristics of a squa-
mous odontogenic tumor lesion that closely resem-
bled the clinical presentation of localized periodontal
disease.
CASE REPORT
A 43-year-old African-American male patient was
referred to The Ohio State University Section of Peri-
odontology by his general dental practitioner for peri-
odontal surgical care. The medical history of the
patient was unremarkable. His dental history revealed
restorative care as well as non-surgical periodontal
therapy for a period of a year prior to examination in
the periodontal graduate clinic. The patient reported
removal of an unerupted tooth from the area of the
upper left canine approximately 20 years earlier. Pre-
vious dental practitioners explained the radiographic
* Section of Periodontology, College of Dentistry, The Ohio State lucency between teeth numbers 11 and 12 as an
University, Columbus OH.
† Section of Oral and Maxillofacial Surgery.
osseous defect which resulted from the surgical
‡ Department of Pharmacology, College of Medicine. removal of the impacted tooth.

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Case Report
Clinical examination revealed gingival recession between teeth numbers 11 and 12. Intrasulcular inci-
on the facial surfaces of teeth numbers 11, 12, and sions were made and a palatal mucoperiosteal flap
13, with probing depths of 7 mm distobuccal and was reflected for access to the lesion. Gently pack-
distolingual to tooth number 11. Transgingival prob- ing ribbon-gauze against the palatal bone, the lesion
ing revealed significant interproximal bone loss was displaced facially while still attached to the buc-
amounting to almost three-quarters of the buccal and cal gingiva (Fig. 3). The neoplasm was subsequently
and one-third of lingual bone between teeth numbers dissected from the buccal mucogingival flap with rel-
11 and 12. ative ease and the lesion enucleated in toto. The lesion
The gingiva exhibited a mild marginal erythema. was firm and rubbery in consistency and no fluid was
There were no noticeable signs of soft tissue swelling aspirated. The roots of the adjacent teeth were root-
or osseous expansion. The papilla in the interdental planed, irrigated with saline, and the mucoperiosteal
region between teeth numbers 11 and 12 had a slight flap apically positioned and closure achieved using
nodular appearance and minor loss of papillary height interrupted 4/0 silk sutures. Due to the osseous defect
was evident (Fig. 1). Bleeding on probing was present observed (horizontal defect with no bony walls),
at the sites around these teeth and both teeth were regeneration of the bone was not considered. The
responsive to electrical stimulation. No sensitivity to surgical site was monitored for 18 months postoper-
percussion was noted for teeth numbers 11 or 12. atively with no sign of a recurrence (Fig. 4). Although
Radiographic assessment showed a radiolucency a mild marginal gingival inflammation persists, all
with severe alveolar bone loss localized between teeth probing depths for teeth numbers 11 and 12 were
numbers 11 and 12, which was associated with less than 4 mm 18 months postoperatively.
markedly diverging roots (Fig. 2). Periapical radi-
Histopathology
ographs from the previous year did not reveal any
Macroscopically, the specimen was a tan-gray, soft
remarkable differences in the size or extent of the
tissue measuring 18 × 12 × 8 mm (Fig. 5). Micro-
current radiolucent finding.
Based on the clinical findings, the differential diag-
nosis included peripheral giant cell lesion, fibro-
osseous lesion, localized severe chronic periodonti-
tis, residual odontogenic cyst, squamous odontogenic
tumor, ameloblastoma, lateral periodontal cyst, and
radicular cyst.
Surgical Treatment
Due to the radiographic appearance, arrangement
was made for a biopsy of the interdental lesion

Figure 1. Figure 2.
Preoperative view of teeth numbers 10 through 13. Periapical radiograph of teeth numbers 11 and 12.

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Case Report

Figure 3. Figure 4.
Neoplasm has been displaced facially while maintaining attachment Postoperative view of teeth numbers 10 through 13 eighteen months
to the buccal mucoperiosteal flap. following surgical excision.

Figure 5. Figure 6.
SOT measuring 18 × 12 × 8 mm. Histophotomicrograph of SOT (hemotoxylin and eosin; original
magnification ×40).

scopically, the sections showed a soft tissue speci-

men, which was consistent of a tumor of epithelial ori- aware of SOT because of its ability to closely mimic
gin within a mature collagenous stroma. The lesional periodontitis. SOT can share many of the character-
cells were characterized by formation of variably sized istic features of periodontitis including gingival inflam-
nests and cords of uniform, benign-appearing, squa- mation, deep probing depths, and radiographic evi-
mous epithelium (Fig. 6). The individual cellular dence of bone loss. The principal method to clinically
islands revealed a lack of peripheral cellular pal- differentiate SOT from periodontitis is the appear-
isading (Fig. 6). Occasional vacuolization and kera- ance of a unilocular, triangle-shaped radiolucency
tinization were noted (Fig. 6). The microscopic diag- that may sometimes also be located between the
nosis was SOT. roots of teeth. There were no clinical signs or symp-
toms associated with teeth numbers 11 and 12 that
DISCUSSION would suggest the presence of SOT except for the
Although an extremely rare lesion of the periodon- incidental finding on radiographic assessment.
tium, periodontists and general dentists should be Histologically, SOT contains multiple, small islands

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Case Report
of squamous epithelium with a moderately cuboidal our case, no sign of a recurrence has been evident
or flattened peripheral basal cell layer in a collage- 18 months postoperatively. Although it is considered
nous fibrous connective tissue stroma. Cystic degen- a benign tumor, recent observations suggest the pos-
eration of the islands has been reported and some sibility of carcinomatous transformation.10 A rare case
have been shown to contain both prekeratin and lam- of intraosseous squamous cell carcinoma arising in
inar calcified masses.1,4 Clear cells show a positive association with and presumed to be a malignant
PAS reaction which indicates the presence of glyco- variant of a SOT lesion emphasizes the need for
gen. The lack of nuclear palisading by the peripheral histopathological assessment of all oral pathology
epithelial cell layer of the tumor islands should help specimens.
rule out the diagnosis of ameloblastoma. Similarly, the
bland histologic features of the epithelial nests should ACKNOWLEDGMENTS
exclude consideration of carcinoma. The authors thank Dr. Charles Solt for his editorial
The histogenesis of SOT remains controversial. review of this manuscript. This study was supported
Some researchers have suggested that these tumors by The Ohio State University Section of Periodontol-
originate from gingival epithelium5 or gingival rests ogy.
of Serres;6 however, most investigators support the
theory that SOT develops from the rests of
REFERENCES
Malassez.1,4,7 Supporting this concept is the obser-
1. Pullon PA, Shafer WG, Elzay RP, Kerr DA, Corio RL.
vation that SOT is often associated with the roots of Squamous odnontogenic tumor: Report of six cases of
erupted vital teeth, although impacted teeth can also a previously undescribed lesion. Oral Pathol 1975;40:
be affected.1 In this case, no expansion of the alve- 616-630.
olar bone or swelling of the gingiva was present. Fur- 2. Leider AS, Jonker AL, Cook HE. Multicentric familial
thermore, the tight adherence of the tumor to the squamous odontogenic tumor. Oral Surg Oral Med Oral
Pathol 1989;68:175-181.
canine root and the ease of separation from the buc- 3. Buchner A, Sciubba JJ. Peripheral epithelial odonto-
cal soft tissues suggest that the origin of the SOT is genic tumors: A review. Oral Surg Oral Med Oral Pathol
most likely the rests of Malassez. The rests of 1987;63:688-697.
Malassez have long been thought as having no func- 4. Doyle JL, Grodjesk JE, Dolinsky HB, Rafel SS. Squa-
tion; however, recently they have been implicated in mous odontogenic tumor: Report of three cases. J Oral
Surg 1977;35:994-996.
having pathologic potential.8,9 The past history of an 5. van der Waal I, de Rijeke TBM, van der Kwast WAM.
extraction from the site of SOT in this case and the Possible squamous odontogenic tumor: Report of a
presence of inflammation may have been triggering case. J Oral Surg 1980;38:460-462.
factors in development of neoplastic growth from 6. Carr RF, Carlton DM, Marks RB. Squamous odonto-
rests of Malassez. genic tumor: Report of a case. J Oral Surg 1981;39:
297-298.
While considered a benign neoplasm, SOT has 7. Goldblatt LL, Brannon RB, Ellis GL. Squamous odon-
been known to infiltrate into adjacent tissues, with togenic tumor: Report of five cases and review of the
resorption of alveolar bone and invasion of the over- literature. Oral Surg Oral Med Oral Pathol 1982;54:187-
lying gingiva and oral mucosa. This is especially true 196.
in the maxilla, where occasionally invasion of adja- 8. Browne RM. The pathogenesis of odontogenic cysts: A
review. J Oral Pathol 1975;4:31-46.
cent structures such as the sinus and nasal cavity 9. Spouge JD. A new look at the rests of Malassez: A
has necessitated wide en bloc resection, including review of their embryological origin, anatomy, and pos-
hemimaxillectomy. As seen in this case, the tumor is sible role in periodontal health and disease. J Peri-
slow-growing and typical conservative surgical inter- odontol 1980;51:437-444.
vention including enucleation and curettage has often 10. Ide F, Shimoyama T, Horie N, Shimizu S. Intraosseous
squamous cell carcinoma arising in association with a
proved curative. It should be noted that, depending squamous odontogenic tumor of the mandible. Oral
on the amount of bony destruction, the possibility for Oncol 1999;35:431-434.
regeneration of the periodontium should be consid-
ered. In this case, the destruction of osseous tissue Correspondence: Dr. Angelo Mariotti, Section of Periodon-
did not provide adequate architecture for regenera- tology, College of Dentistry, P.O. Box 182357, The Ohio
State University, 305 W. 12th Avenue, Columbus, OH
tion to be considered. In the original description of the 43218-2357.
lesion by Pullon et al.1 only one case had a recurrence
which required more extensive surgical excision. In Accepted for publication January 14, 2002.

656 Neoplasms in the Periodontium Volume 73 • Number 6