Sporadic Odontogenic Keratocyst With Extensive Involvement of Maxillary Antra, Impacted Teeth and Exhibiting a Focus of Ameloblastoma: Case Series and Review of Literature

Bernard Lam DDS, MD, OMFS, Thomas Jefferson University Hospital, Philadelphia, PA
Daniel Taub DDS, MD, Oral and Maxillofacial Surgery, Thomas Jefferson University Hospital, Philadelphia, PA
Shachika Khanna BDS, DMD, Oral & Maxillofacial Surgery, Thomas Jefferson University Hospital, Philadelphia, PA
Lionel Gold DDS, Oral and Maxillofacial Surgery, Thomas Jefferson University Hospital, Philadelphia, PA

Sporadic odontogenic keratocyst with extensive involvement of maxillary antra, impacted teeth and exhibiting a focus of ameloblastoma: Case series and review of literature.

An odontogenic keratocyst (OKC) is a persistent, benign lesion that is currently identified by many as a neoplastic, or quasi-neoplastic entity. The cystic lesion arises from the primitive odontogenic lamina. The OKC was first described by Phillipsen in 1956, and is most commonly found in the mandibular rami and molar areas.

OKC is not commonly found in the maxillary antra involving impacted third molars and rarely occurs in synchronous with ameloblastoma. A review of literature exhibits only a few cases that involve the antra, and even more seldom seen with ameloblastoma. In this case series, we relate multiple cases of OKC that involves the maxilla, antra, and molar teeth that appears to be sporadic and not syndromic.  One case exhibits OKC involvement of the maxilla and antra with a recognizable focus of ameloblastoma in an adolescent. 

Each case was treated with enucleation of the cyst followed by chemical curettage with carnoy's solution. 

Definitive diagnosis relies on accurate histopatholgic, radiographic, and clinical examination in order to provide proper treatment with avoidance of recurrence.  Based on the extent of the lesions, and high recurrence rate of OKC, enucleation of cysts with resection without continuity defect followed by Carnoy's solution and close follow up was the treatment of choice.  However, the obvious question is unanswered in regards to the lesion with the focus of ameloblastoma; if the lesion was not treated, would an exuberant ameloblastoma resulted?

References:

1: Siar CH, and NG KH. Combined ameloblastoma and odontogenic keratocyst or keratinizing ameloblastoma. British Journal of Oral and Maxillofacial Surgery. 1993 31, 183-186.

2. Byun JH, Kang YH, Choi MJ, Park BW. Expansile keratocystic odontogenic tumor in the maxilla: immunohistochemical studies and review of literature. J Korean Assoc Oral Maxillofac Surg. 2013 39, 182-187.

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