Journal of Cranio-Maxillo-Facial Surgery 40 (2012) e355ee357

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Case report

Squamous cell carcinoma arising in dentigerous cysts

Serryth Colbert a, *, Peter A. Brennan a, Jeffery Theaker b, Barrie Evans c
a

Department of Oral and Maxillofacial Surgery, Queen Alexandra Hospital, Portsmouth, UK

Department of Histopathology, Southampton General Hospital, UK
c
Department of Oral and Maxillofacial Surgery, Southampton General Hospital, UK
b

a r t i c l e i n f o

a b s t r a c t

Article history:
Paper received 8 February 2011
Accepted 31 January 2012

We present two cases of squamous cell carcinoma (SCC) arising in dentigerous cysts. Malignant transformation in dentigerous cysts is rare giving rise to diagnostic difculties. We propose imaging at an
early stage to reduce delays in diagnosis.
2012 European Association for Cranio-Maxillo-Facial Surgery.

Keywords:
Squamous cell carcinoma
Dentigerous cyst
Malignant transformation

1. Introduction
Dentigerous cysts are common e 1.44 per 100 unerupted teeth
(Mourshed, 1964). SCCs arising in these lesions are rare e the
estimated incidence is 1e2 per 1000 (Stoelinga and Bronkhorst,
1988). The diagnosis is usually made in retrospect due to lack of
symptoms and radiographic changes. Routine cone-beam scanning
of all dentigerous cysts to improve the preoperative diagnosis is
unpractical and will rarely detect malignant change.
1.1. Aims
We present two cases of SCC arising in dentigerous cysts with
differing outcomes e one conned to the cyst lining itself and
the other with extensive soft tissue invasion. We advocate early
imaging and enucleation of these cysts to minimise delays in
diagnosis.
2. Case Report
2.1. Case Report 1
A 66-year-old male presented with bilateral unilocular radiolucencies associated with unerupted and impacted lower third
molars. The presumptive diagnosis was bilateral dentigerous cysts
(Fig. 1). Following enucleation, histology conrmed this (Fig. 2),

* Corresponding author. Department of Oral & Maxillofacial Surgery, Queen

Alexandra Hospital, Portsmouth PO6 3LY, UK. Tel.: 44 (0) 2392286000.
E-mail address: serryth@yahoo.com (S. Colbert).

with however, invasive squamous cell carcinoma (Fig. 3) arising

from the cyst lining on the left. The lesion was excised and the
defect reconstructed with a composite bular free ap followed by
adjuvant chemo-radiotherapy. The patient is currently 3 years post
resection and reconstruction and tumour free.
2.2. Case Report 2
A 42-year-old man presented with 5-year history of recurrent
infections in a cystic lesion associated with an impacted lower right
second premolar. The patient reported slight numbness of the
lower lip. A decision was made to extract the impacted lower right
second premolar and enucleate the associated cystic lesion.
However, at operation the lesion had extended into the adjacent
soft tissues and measured approximately 1.5 cm in diameter.
Histology of the lesion conrmed malignant change in the dentigerous cyst, inltrating the body of the mandible and extending
laterally through the bone to involve the soft tissues and subcutaneous tissues. In retrospect, given the histology, a biopsy would
have been the preferred initial treatment. He was treated with right
mandibular resection, neck dissection and reconstruction with
a free bular ap followed by adjuvant radiotherapy. The tumour
recurred within a year and the patient developed an extensive
fungating carcinoma before succumbing to the disease (Fig. 4).
3. Discussion
Dentigerous cysts are the most common odontogenic cysts and
are associated with unerupted/impacted teeth. They occur most
frequently in the mandibular third molar, maxillary canine,

1010-5182/$ e see front matter 2012 European Association for Cranio-Maxillo-Facial Surgery.
doi:10.1016/j.jcms.2012.01.020

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S. Colbert et al. / Journal of Cranio-Maxillo-Facial Surgery 40 (2012) e355ee357

Fig. 1. Bilateral mandibular angle radiolucencies associated with unerupted wisdom

teeth without atypical imaging features suggestive of malignant transformation.

Fig. 4. Fungating carcinoma with extensive soft tissue inltration beyond the body of
the mandible (arrow).

Fig. 2. Histology of dentigerous cyst showing epithelial lining (up arrow) separating
cystic content (down arrow) from the surrounding stroma (curved arrow).

Fig. 3. Invasive keratinising squamous cell carcinoma within the dentigerous cyst (arrow).

maxillary third molar, and mandibular second premolar regions.

Their incidence is estimated at 1.44 per 100 unerupted teeth
(Mourshed, 1964). Carcinoma arising in dentigerous cysts is rare,
with a reported incidence of 1 to 2/1000 (Norris et al., 1984). Dentigerous cysts are diagnosed between the second and third decades;

by contrast, the mean age for SCC in these cysts is 64 years suggesting they develop slowly (Maxymiw and Wood, 1991). Eightytwo cases have been reported in the English literature (Chaisuparat
et al., 2006). Over the past 3 years, there has only been one report of
malignant transformation in an odontogenic cyst, published in the
Journal of Cranio-Maxillofacial Surgery (Yamanda et al., 2009).
Evidence suggests that transformation from normal epithelial
cyst lining to SCC is due to chronic inammation (Browne & Gough,
1972). Malignancy in the cyst wall is usually unexpected at the time
of presentation and the diagnosis is usually made following
enucleation (Pearcey, 1985). Dentigerous cysts are usually diagnosed on plain radiographs. Jagged or irregular margins with
indentations and indistinct borders are said to suggest possible
malignant change (Chaisuparat et al., 2006). The malignant transformation is a slow process and can take 20e30 years. Johnson et al.
advise extracting third molars with signicant radiographic
changes in cyst size >2.5 mm (Johnson et al., 1994). Cavalcanti
MG et al. claim that cone beam imaging has increased early
detection rate of carcinomatous changes, because of its sensitivity
in establishing the nature of the lesion (Cavalcanti et al., 2005). In
practice tomographic imaging is limited to larger lesions and those
that may encroach on the ID bundle.
Glosser and Campbell showed that the incidence of dentigerous
cysts associated with impacted third molar teeth is higher than
reported in radiographic studies alone (Glosser and Campbell,
1999). Current practice is to investigate for dentigerous cysts with
one dimensional plain radiographs. The advent of cone beam
imaging has increased the detection rate of this condition, but
cost implications would preclude its use routinely. In practice,
computed tomographic imaging is only performed if cystic lesions
encroach on the inferior dental bundle with evidence of neurological involvement. Following current practice, malignant change
in dentigerous cysts will continue to be a diagnostic difculty being
made in retrospect after the lesion has been excised and meticulously examined by the pathologist. Therefore, we advocate early
imaging and removal of impacted teeth associated with dentigerous cysts as the treatment of choice.
4. Conclusion
These cases clearly demonstrate the importance of clinician
awareness of the malignant potential of apparently innocuous
cystic lesions. They also underscore the importance of a careful

S. Colbert et al. / Journal of Cranio-Maxillo-Facial Surgery 40 (2012) e355ee357

histological examination and the necessity of obtaining a biopsy

to prevent a misdiagnosis of such cystic lesions. As malignant
transformation is rare and cross-sectional imaging unlikely to
become routinely used in most dentigerous cysts, it is our view that
retrospective diagnosis is likely to remain the norm in the majority
of cases. The clinicians index of suspicion of malignant transformation should be raised in older patients, presentation with
altered sensation and atypical radiographic ndings. Early diagnosis and early surgical removal with sufcient margins of safety is
crucial when treating these rare tumours. Therefore, we suggest
early imaging and removal of impacted teeth associated with
dentigerous cysts.
Conict of interest
None.
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