Ectodermal dysplasia (ED) is a congenital syndrome characterized chiefly by abnormalities of tissues of ectodermal origin, namely skin, nails, hair, and teeth. Dental treatment of patients with ED is necessary because it affords the opportunity to develop normal forms of speech, chewing, swallowing, and normal facial support. Because there are few reports focusing on implants inserted in bone grafted in patients with ED, we therefore performed a retrospective study on 44 implants inserted in 4 patients to detect those variables acting on survival and crestal bone remodeling around implant neck in such subjects. Forty-four fixtures were analyzed. Several patient-related (age and sex), anatomic (maxilla and mandible and tooth site), implant (type, length, and diameter), surgical (sites and types of grafts), and prosthetic (type of loading) variables were investigated. Implant failure and peri-implant bone resorption were considered as predictors of clinical outcome. Kaplan-Meier algorithm and Cox regression were then performed to detect those variables statistically associated with the clinical outcome. Implant length and diameter ranged from 11.5 to 15 mm and from 3.5 to 4.0 mm, respectively. Implants were inserted to replace 12 incisors, 12 cuspids, 11 premolars, and 9 molars. No implant was lost. On the contrary, implant's length, grafted sites, and type of loading affected univariate analysis, but these data were not confirmed by multivariate algorithm. Dental implants and bone grafts to orally rehabilitate patients with ED are valuable devices with no difference if compared with unaffected patients, at least in adults.