Review
Dental implants in patients with ectodermal dysplasia: A systematic review

https://doi.org/10.1016/j.jcms.2018.05.038Get rights and content

Highlights

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    Review of implants in ectodermal dysplasia patients: 1472 implants, 228 patients.

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    72/1391 implants (5.2%) failed; 20-year cumulative survival rate: 84.6%.

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    24.6% of implants placed in children (0–17 years of age), 7% failure rate.

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    Probability of failure: 4.5% (95%CI 3.5%–5.6%, p < 0.001).

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    Implants in infant/adult ectodermal dysplasia patients present a high survival rate.

Abstract

Purpose

This study sought to assess the clinical outcome and survival rate of oral implants placed in individuals with ectodermal dysplasia (ED), based on previously published studies.

Methods

An electronic search without time restrictions was undertaken in 5 databases (PubMed/Medline, Web of Science, ScienceDirect, J-Stage, Lilacs). Descriptive statistics, Kaplan Meier estimator and implant failure probability were calculated.

Results

90 publications were included, reporting 228 ED patients that received 1472 implants (1392 conventional, 47 zygomatic, 33 mini-implants). Mean age of the patients was 20.2 ± 6.8 years (2–56). Patients had a mean of 3.2 ± 2.5 maxillary and 2.1 ± 2.6 mandibular permanent teeth (min–max, 0–14). Patients received a mean of 8.2 ± 3.8 implants (1–20). Most implants were placed in the third decade of life, 24.6% of the implants were placed in children (0–17 years of age). 1391 implants had information on follow-up (72 failures, 5.2%). The 20-year CSR was 84.6%. The probability of failure was 4.5% (95%CI 3.5%–5.6%, p < 0.001). Additional treatments performed were Le Fort I (99 implants, 20 patients, 3.5% failed), grafting (497 implants, 77 patients, 5.2% failed), distraction osteogenesis (79 implants, 16 patients, 10.1% failed). Mean follow-up was 42.9 ± 41.9 months (min–max, 2–240).

Conclusions

Dental implants placed in ED patients, either infants or adults, present a high survival rate (20-year CSR 84.6%).

Introduction

Ectodermal dysplasia (ED) encompasses a number of genetic syndromes characterized by a congenital defect in two or more of the ectodermal structures of the body. The condition is estimated to occur in approximately 1 in 100,000 live births, and approximately 132 different hereditary syndromes related to ED have been identified (Clarke, 1987). The syndromes usually affect the hair, teeth, nails, sweat glands, craniofacial structures, digits, and occasionally mesodermal abnormalities (Clarke, 1987). The impact on the oral and maxillofacial region includes decreased growth of the mandible and maxilla, deficient development of the maxillary and mandibular alveolar ridges, significant reduction in salivary secretions, and malformations and anomalies of number and shape of primary and permanent teeth (Martin et al., 2005).

As many of these patients present oligodontia (absence of 6 or more teeth) or anodontia (complete absence of teeth), a prosthetic rehabilitation is usually desirable. The degree of dentoalveolar tissue deficiency can make an implant-supported prosthesis an appropriate method of definitive occlusal restoration in these patients. However, as the absence of teeth is congenital, this raises the issue of placement of oral implants in growing children, mainly due to the influence of craniofacial growth on the implant's behavior (Singer et al., 2012). The aim of the present review was to assess the clinical outcome and survival rate of oral implants used for the oral rehabilitation of ED patients.

Section snippets

Materials and methods

This study followed the PRISMA Statement guidelines (Moher et al., 2009).

Literature search

The study selection process is summarized in Fig. 1. The search strategy in the databases resulted in 991 papers. Search in Google Scholar resulted in 5 eligible papers not found in the five main databases. A number of 162 articles were cited in more than one database (duplicates). The reviewers independently screened the abstracts for those articles related to the aim of the review. Of the resulted 834 studies, 720 were excluded for not being related to the topic or not presenting clinical

Discussion

The oral rehabilitation of patients with implants is generally delayed until the cessation of growth because an implant does not exhibit dentoalveolar adaptation in response to vertical alveolar growth or local bony remodeling as would occur in the case of a tooth (Björk and Skieller, 1972, Thilander et al., 1992). However, the use of removable dentures in the deficient residual basal bone structures usually observed in individuals with ED could be a cause of functional and psychological

Conclusions

Dental implants placed in ED patients, either infants or adults, present a high survival rate (20-year CSR 84.6%).

Funding/grant support

This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

Declaration of conflicting interests

There are no conflicts of interest to declare.

Acknowledgements

We would like to thank Dr. Hussein El Charkawi and Dr. Sanjeev Deshpande, who provided us some additional information, and Dr. Bruno Salles Sotto-Maior, who provided us his article. Last but not least, we would like to thank the librarians of Malmö University (with a special thanks to Ms. Anneli Svensson), who helped us to obtain some articles.

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© 2018 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.