Ectodermal dysplasias: Classification and organization by phenotype, genotype and molecular pathway.


Journal

American journal of medical genetics. Part A
ISSN: 1552-4833
Titre abrégé: Am J Med Genet A
Pays: United States
ID NLM: 101235741

Informations de publication

Date de publication:
03 2019
Historique:
received: 29 10 2018
revised: 19 12 2018
accepted: 21 12 2018
pubmed: 1 2 2019
medline: 17 4 2020
entrez: 1 2 2019
Statut: ppublish

Résumé

An international advisory group met at the National Institutes of Health in Bethesda, Maryland in 2017, to discuss a new classification system for the ectodermal dysplasias (EDs) that would integrate both clinical and molecular information. We propose the following, a working definition of the EDs building on previous classification systems and incorporating current approaches to diagnosis: EDs are genetic conditions affecting the development and/or homeostasis of two or more ectodermal derivatives, including hair, teeth, nails, and certain glands. Genetic variations in genes known to be associated with EDs that affect only one derivative of the ectoderm (attenuated phenotype) will be grouped as non-syndromic traits of the causative gene (e.g., non-syndromic hypodontia or missing teeth associated with pathogenic variants of EDA "ectodysplasin"). Information for categorization and cataloging includes the phenotypic features, Online Mendelian Inheritance in Man number, mode of inheritance, genetic alteration, major developmental pathways involved (e.g., EDA, WNT "wingless-type," TP63 "tumor protein p63") or the components of complex molecular structures (e.g., connexins, keratins, cadherins).

Identifiants

pubmed: 30703280
doi: 10.1002/ajmg.a.61045
pmc: PMC6421567
mid: NIHMS1013279
doi:

Substances chimiques

Biomarkers 0

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

442-447

Subventions

Organisme : NIDCR NIH HHS
ID : R01 DE016079
Pays : United States
Organisme : NIDCR NIH HHS
ID : R13 DE018845
Pays : United States

Informations de copyright

© 2019 Wiley Periodicals, Inc.

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Auteurs

John Timothy Wright (JT)

Department of Pediatric Dentistry, Bauer Hall CB#7450, School of Dentistry, University of North Carolina, Chapel Hill, North Carolina.

Mary Fete (M)

National Foundation for Ectodermal Dysplasias, Fairview Heights, Illinois.

Holm Schneider (H)

Department of Pediatrics, University Hospital Erlangen, Erlangen, Germany.

Madelaine Zinser (M)

National Foundation for Ectodermal Dysplasias, Fairview Heights, Illinois.

Maranke I Koster (MI)

NFED Scientific Advisory Council, Fairview Heights, Illinois.
Dermatology and Ophthalmology, University of Colorado Anschutz Medical Campus, Aurora, Colorado.

Angus J Clarke (AJ)

Cancer & Genetics, School of Medicine, Cardiff University, Wales, United Kingdom.

Smail Hadj-Rabia (S)

Department of Dermatology, Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC), INSERM U1163, Descartes - Sorbonne Paris Cité University, Imagine Institute, Necker-Enfants Malades Universitary Hospital, Paris, France.

Gianluca Tadini (G)

Center for Inherited Cutaneous Diseases, Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Foundation IRCCS Ca' Granda, Ospedale Maggiore Policlinico, Milan, Italy.

Nina Pagnan (N)

Department of Genetics, Federal University of Parana, Curitiba, Brazil.

Atila F Visinoni (AF)

Positivo University, Curitiba, Brazil.

Birgitta Bergendal (B)

National Oral Disability Center for Rare Disorders, The Institute for Postgraduate Dental Education, Jönköping, Sweden.

Becky Abbott (B)

NFED for Treatment & Research, Fairview Heights, Illinois.

Timothy Fete (T)

NFED Scientific Advisory Council, Fairview Heights, Illinois.
Department of Child Health, University of Missouri, Columbia, Missouri.

Clark Stanford (C)

NFED Scientific Advisory Council, Fairview Heights, Illinois.
University of Illinois at Chicago College of Dentistry, Chicago, Illinois.

Clayton Butcher (C)

Departments of Medicine and Child Health, University of Missouri School of Medicine, Columbia, Missouri.

Rena N D'Souza (RN)

Academic Affairs and Education, Health Sciences, The University of Utah, Salt Lake City, Utah.

Virginia P Sybert (VP)

Division of Medical Genetics, Department of Medicine, University of Washington School of Medicine, Seattle, Washington.

Maria I Morasso (MI)

Laboratory of Skin Biology, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.

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