Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review

Abstract Objective The objective of this systematic review was to determine the orthodontic and dentofacial orthopedic treatments carried out in patients with ectodermal dysplasia to facilitate functional and aesthetic rehabilitation. Methods The systematic review was conducted in accordance with th...

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Main Authors: Marina Cerezo-Cayuelas, Amparo Pérez-Silva, Clara Serna-Muñoz, Ascensión Vicente, Yolanda Martínez-Beneyto, Inmaculada Cabello-Malagón, Antonio José Ortiz-Ruiz
Format: Article
Language:English
Published: BMC 2022-10-01
Series:Orphanet Journal of Rare Diseases
Subjects:
Online Access:https://doi.org/10.1186/s13023-022-02533-0
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author Marina Cerezo-Cayuelas
Amparo Pérez-Silva
Clara Serna-Muñoz
Ascensión Vicente
Yolanda Martínez-Beneyto
Inmaculada Cabello-Malagón
Antonio José Ortiz-Ruiz
author_facet Marina Cerezo-Cayuelas
Amparo Pérez-Silva
Clara Serna-Muñoz
Ascensión Vicente
Yolanda Martínez-Beneyto
Inmaculada Cabello-Malagón
Antonio José Ortiz-Ruiz
author_sort Marina Cerezo-Cayuelas
collection DOAJ
description Abstract Objective The objective of this systematic review was to determine the orthodontic and dentofacial orthopedic treatments carried out in patients with ectodermal dysplasia to facilitate functional and aesthetic rehabilitation. Methods The systematic review was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-analysis statement. We systematically searched PubMed, Web of Science, Scopus, Scielo, LILACS, EBSCOhost and Embase databases up to 6 January 2022. We included articles describing patients with any type of ectodermal dysplasia who received orthodontic or dentofacial orthopedic treatment to facilitate functional and aesthetic oral rehabilitation. The search was not restricted by language or year of publication. The quality of the studies was assessed using the Joanna Briggs Institute Quality Assessment Scale of the University of Adelaide for case series and case reports. The review was registered at the University of York Centre for reviews (CRD42021288030). Results Of the initial 403 studies found, 29 met the inclusion criteria. After applying the quality scale, 23 were left for review—21 case reports and 2 case series. The initial age of patients ranged from 34 months to 24 years. Thirteen studies were on hypohidrotic and/or anhidrotic ectodermal dysplasia, of which two were X-chromosome linked. In one study, the patient had Wiktop syndrome, and in nine the type of ectodermal dysplasia was not specified. The duration of treatment was 7 weeks to 10 years. The treatments described were: fixed orthodontic appliances or simple acrylic plates designed for tooth movement, including leveling and aligning, closing of diastemata, retraction of impacted teeth in the dental arch; clear aligners; fixed and/or removable appliances for the correction of skeletal and/or dentoalveolar relationships; palatal expanders in combination with face masks for orthopedic traction of the maxilla; and orthognathic surgery. Only three studies provided cephalometric data. Conclusion The level of evidence of the articles reviewed was low and most orthopedic and dentofacial orthodontic treatments described were focused on correcting dental malpositioning and jaw asymmetries and not on stimulating growth from an early age. Studies with greater scientific evidence are needed to determine the best treatment for these patients.
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spelling doaj.art-777e31c2611f452e858143f8cdd7e64e2022-12-22T04:37:01ZengBMCOrphanet Journal of Rare Diseases1750-11722022-10-0117111810.1186/s13023-022-02533-0Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic reviewMarina Cerezo-Cayuelas0Amparo Pérez-Silva1Clara Serna-Muñoz2Ascensión Vicente3Yolanda Martínez-Beneyto4Inmaculada Cabello-Malagón5Antonio José Ortiz-Ruiz6Department of Integrated Pediatric Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, Morales Meseguer Hospital, University of MurciaDepartment of Integrated Pediatric Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, Morales Meseguer Hospital, University of MurciaDepartment of Integrated Pediatric Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, Morales Meseguer Hospital, University of MurciaOrthodontics Unit, University Dental Clinic, Biomedical Research Institute of Murcia, University of MurciaDepartment of Preventive and Community Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, University of MurciaDepartment of Integrated Pediatric Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, Morales Meseguer Hospital, University of MurciaDepartment of Integrated Pediatric Dentistry, School of Dentistry, Biomedical Research Institute of Murcia, Morales Meseguer Hospital, University of MurciaAbstract Objective The objective of this systematic review was to determine the orthodontic and dentofacial orthopedic treatments carried out in patients with ectodermal dysplasia to facilitate functional and aesthetic rehabilitation. Methods The systematic review was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-analysis statement. We systematically searched PubMed, Web of Science, Scopus, Scielo, LILACS, EBSCOhost and Embase databases up to 6 January 2022. We included articles describing patients with any type of ectodermal dysplasia who received orthodontic or dentofacial orthopedic treatment to facilitate functional and aesthetic oral rehabilitation. The search was not restricted by language or year of publication. The quality of the studies was assessed using the Joanna Briggs Institute Quality Assessment Scale of the University of Adelaide for case series and case reports. The review was registered at the University of York Centre for reviews (CRD42021288030). Results Of the initial 403 studies found, 29 met the inclusion criteria. After applying the quality scale, 23 were left for review—21 case reports and 2 case series. The initial age of patients ranged from 34 months to 24 years. Thirteen studies were on hypohidrotic and/or anhidrotic ectodermal dysplasia, of which two were X-chromosome linked. In one study, the patient had Wiktop syndrome, and in nine the type of ectodermal dysplasia was not specified. The duration of treatment was 7 weeks to 10 years. The treatments described were: fixed orthodontic appliances or simple acrylic plates designed for tooth movement, including leveling and aligning, closing of diastemata, retraction of impacted teeth in the dental arch; clear aligners; fixed and/or removable appliances for the correction of skeletal and/or dentoalveolar relationships; palatal expanders in combination with face masks for orthopedic traction of the maxilla; and orthognathic surgery. Only three studies provided cephalometric data. Conclusion The level of evidence of the articles reviewed was low and most orthopedic and dentofacial orthodontic treatments described were focused on correcting dental malpositioning and jaw asymmetries and not on stimulating growth from an early age. Studies with greater scientific evidence are needed to determine the best treatment for these patients.https://doi.org/10.1186/s13023-022-02533-0Ectodermal dysplasiaOrthodontic treatmentDentofacial orthopedic treatmentAnhidrotic ectodermal dysplasiaHidrotic ectodermal dysplasiaHypohidrotic ectodermal dysplasia
spellingShingle Marina Cerezo-Cayuelas
Amparo Pérez-Silva
Clara Serna-Muñoz
Ascensión Vicente
Yolanda Martínez-Beneyto
Inmaculada Cabello-Malagón
Antonio José Ortiz-Ruiz
Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
Orphanet Journal of Rare Diseases
Ectodermal dysplasia
Orthodontic treatment
Dentofacial orthopedic treatment
Anhidrotic ectodermal dysplasia
Hidrotic ectodermal dysplasia
Hypohidrotic ectodermal dysplasia
title Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
title_full Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
title_fullStr Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
title_full_unstemmed Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
title_short Orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia: a systematic review
title_sort orthodontic and dentofacial orthopedic treatments in patients with ectodermal dysplasia a systematic review
topic Ectodermal dysplasia
Orthodontic treatment
Dentofacial orthopedic treatment
Anhidrotic ectodermal dysplasia
Hidrotic ectodermal dysplasia
Hypohidrotic ectodermal dysplasia
url https://doi.org/10.1186/s13023-022-02533-0
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