Odontogenic cysts are pathological cavities filled with fluid or semifluid material that arise from tissues involved in odontogenesis. The pathogenesis of odontogenic cysts is, however, still uncertain. This study aims to uncover some of the possible mechanisms of odontogenic cyst formation focusing on the role of primary cilia. Primary cilia are organelles located in nearly every mammalian cell, which mediate communication between cells and are necessary for the transportation of signal molecules. Mutations in ciliary-associated proteins result in a group of diseases known as ciliopathies, which are often associated with cystic growth in various organs (e.g. kidneys, liver, lungs). Therefore, we decided to investigate the role of primary cilia in odontogenic cysts and to find a potential connection between ciliary dysfunction and the development of odontogenic cysts. Here, we analysed changes in the expression of ciliary genes between follicular cysts and odontogenic keratocysts, while using follicular sack as control tissue. We focused on Ift genes responsible for retrograde and anterograde transport in cilia. We also evaluated the morphology of primary cilia in the epithelium of odontogenic cysts using immunofluorescence. Furthermore, we determined the levels of proliferation and apoptosis as well as overall tissue histology in different types of oral cysts. Besides the most common follicular cysts and odontogenic keratocysts, we also described a specific case report of a patient with multiple cysts in the oral cavity. Understanding the genetic background and the role of primary cilia in the development of odontogenic cysts may uncover some general processes of ciliopathic cystogenesis, as well as it can lead in the future to new therapeutic venues for this possibly debilitating disease. Acknowledgement: This work was supported by the Ministry of Health (grant NU20-08-00205).