Patients with Down syndrome may not be cooperative during dental treatment and, in cases of complex treatment, it is necessary to use oral or inhaled sedation and/or general anesthesia, which represent safe and efficient treatment resources. There are few reports about oral rehabilitation with dental implants in patients with Down syndrome in the literature. This paper presents the case of a patient with Down syndrome and moderate mental retardation who suffered avulsion of the maxillary left central incisor after a fall, and was treated by means of a single dental implant with immediate load carried out under general anesthesia. After 4 years of follow up, there has been improvement in the sleep obstructive apnea syndrome due to the use of a tongue crib adapted for providing esthetics and preserving the implant placed.