BACKGROUND: The adenomatoid odontogenic tumor (AOT) is a quite rare odontogenic tumor, with an incidence rate of approximately 12 cases/year worldwide. Attenuated familial adenomatous polyposis (aFAP) is a syndrome characterized by a significant risk to develop colon cancer. The aim of the paper is to describe a case never reported before in the literature: an AOT developed in a patient with aFAP; moreover, we want to show how it appears 5 years after surgery and after the regeneration of the eroded bone tissue, using the Platelet-Rich Fibrin (PRF) as filling material.
CASE PRESENTATION: We report the case of a female 18 years old patient, affected by aFAP; she comes to us with a swelling on the right hemi-face. We performed several radiological exams, and they showed a neoformation approximately 2 cm in diameter: this neoformation packed the upper right canine, therefore, we hypothesized a dentigerous cyst.
We decided to proceed to open biopsy and enucleation of the lesion. An intra-operative endodontic treatment on the adjacent partially resorbed teeth was also performed. Finally, we performed a reconstruction of eroded bone tissue, by use of Platelet-Rich Fibrin as filling material.
The samples fixed and embedded in paraffin have led to the diagnosis of AOT. After 5 years from the surgery, we did not find any clear sign of relapse, in addition, the use of PRF has favored an optimal osteogenesis at the surgical site.
CONCLUSIONS: Undoubtedly, a correct diagnosis of AOT allows to have a more performing clinical and surgical approach. Furthermore, this case could document a new manifestation of aFAP in extra-intestinal site.
The onset of an AOT is quite rare in the general population, and this rarity could represent a critical point for its diagnosis; AOT onset in a patient with aFAP is a finding that could represent a new element of diagnosis and, therefore, the starting point to perform a more effective therapy.
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To cite this article
M. Marrelli, A. Pacifici, G. Di Giorgio, M. Cassetta, L.V. Stefanelli, M. Gargari, L. Promenzio, S. Annibali, M.P. Cristalli, E. Chiaravalloti, L. Pacifici, M. Tatullo
Diagnosis and treatment of a rare case of adenomatoid odontogenic tumor in a young patient affected by attenuated familial adenomatosis polyposis (aFAP): case report and 5 year follow-up
Eur Rev Med Pharmacol Sci
Vol. 18 - N. 2