The purpose of this case report is to describe the treatment outcome of a 4-year-old girl with a dentigerous cyst on the primary mandibular right second premolar of her caries-free mouth. Her primary predecessor was extracted and the lesion decompressed by using a removable appliance with a resin projection. The succeeding premolar had not developed further 8 months later, however, and had to be extracted. Crown formation of the succeeding premolar might have been interrupted by the presence of the cyst. To our knowledge, this is the earliest reported asymptomatic dentigerous cyst in the mandibular premolar region with a vital primary second molar.