Use of endosseous implants in a 3-year-old child with ectodermal dysplasia: case report and 5-year follow-up.

The ectodermal dysplasias represent a group of inherited disorders characterized by defects in tissues that are derived from ectoderm. Freire-Maia and Pinheiro describe more than 100 different taxonomic groupings of ectodermal dysplasia across a wide spectrum of clinical presentation. The classic form of ectodermal dysplasia (Christ-Siemens-Touraine syndrome) is thought to be X-linked and involves hypodontia, hypohidrosis, hypotrichosis, and a characteristic facies. Because these individuals don't sweat, this condition is sometimes referred to as X-linked, hypohidrotic ectodermal dysplasia (XLHED). The dental literature describes many conventional prosthetic approaches to the clinical management of these patients." Osseointegration is well documented as a safe and predictable method of tooth replacement.The early clinical research was done in edentulous adults and subsequent studies have confirmed the successful application of this modality in a variety of clinical situations.Almost all of this scientific investigation, however, has been performed in adults, when the dynamics of growth and development are not an issue. Osseointegrated implants are in direct apposition to bone and lack the compensatory mechanism of a periodontal ligament. Research models indicate that osseointegrated fixtures do not move with the growth of the jaws and suggest caution in their use in individuals where growth is incomplete.However, the lack of relevant long-term clinical studies has not prevented clinicians from using implant-assisted prostheses in children. The literature contains several anecdotal reports of the use of dental implants in children, many with anodontia or severe hypodontia, often associated with ectodermal dysplasia, or from trauma.' The purpose of this paper is to present a case report of implant placement in a patient 3 years and 3 months of age, subsequent prosthodontic treatment and 5-year follow-up. We believe this to be the youngest patient documented to have been treated with a dental implant supported dental prosthesis. Case report History A 3-year-old white male diagnosed with Christ-Siemens-Touraine ectodermal dysplasia was referred to the NIDR dental clinic for prosthetic evaluation following implant placement (Fig 1). The implants had been placed by an oral and maxillofacial surgeon in private practice. The patient's mother reported that the rationale for and possible complications of implant placement in a 3-year-old child were not discussed with her prior to placing the implants. The patient was referred to the National Institute of Dental Research (NIDR) by the National Foundation for Ectodermal Dysplasia* for possible participation in an ongoing protocol. The patient was 3 years and 7 months old at presentation and had four mandibular and two maxillary IMZ press fit (Interpore International, Irvine, CA), endosseous implants, which had been placed at age 3 years 3 months. Hydroxyapatite of unknown type was also placed to augment both the maxilla and mandible and is readily seen on radiographs (Fig 2). This patient had only two tooth buds, both located in the maxilla, and had never worn a dental prosthesis. Since the patient had already received implants prior to presenting at NIDR, he was not eligible for the implant protocol. However, beFig 1. Patient with X-linked hypohydrotic ectodermal dysplasia and severe hypodontia at 3 years 7 months. Endosseous dental implants were placed 4 months earlier.