Ataide DSM, Preto KA, dos Santos GL, Costa BE, Kawakami RY, Oliveira DT. How uncommon is the occurrence of orthokeratinized odontogenic cyst in the maxilla? A systematic review and a new case report. J Clin Exp Dent. 2026;18(3):e394-401.

 

doi:10.4317/jced.63735

https://doi.org/10.4317/jced.63735

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