Aims: The main goal of the following case report was to shed the light on the importance of thorough clinical, radiological and histological examinations in order to elaborate a final diagnosis of asymptomatic dentigerous cysts detected in unusual locations. Presentation of Case: A case of dentigerous cyst was identified accidentally in the maxillary left premolar region of an asymptomatic 14-year-old female post an orthodontic consultation. Histological examination of the tissue specimens following enucleation confirmed the diagnosis of a dentigerous cyst. Discussion: Dentigerous cysts are the second most common odontogenic cysts after radicular cysts. They involve impacted, un-erupted, permanent, supernumerary, odontomas and rarely deciduous teeth. Dentigerous cysts are usually painless but may cause facial swelling and delayed tooth eruption. Extensive maxillary involvement and childhood presentation are rare. Radio-graphic and histological examinations should be done to confirm the diagnosis of a dentigerous cyst. Conclusion: In our case, we showed the presence of a maxillary premolar dentigerous cyst that was removed by enucleation. The presence of dentigerous cyst is not always associated with a syndrome and its removal is very important to avoid future complications.