Background: Squamous cell carcinoma arising in a dentigerous cyst is e
xtremely uncommon. The diagnosis of carcinoma arising in a dentigerous
cyst requires microscopically that an area of benign cystic epitheliu
m is observed to transition into squamous cell carcinoma. In the prese
nt case, the patient was diagnosed with squamous cell carcinoma of the
neck with an unknown primary. The primary site was found to be the fo
llicular space associated with the right mandibular third molar. Metho
ds. Light microscopic studies were performed to arrive al the final di
agnosis. Results. Transition of the epithelial lining of the dentigero
us cyst to squamous cell carcinoma was observed in multiple fields. Co
nclusions. The present case demonstrates that odontogenic cysts have t
o be considered a possible source of metastatic disease. (C) 1997 John
Wiley & Sons, Inc.