High penetrance of EDA pathogenic variants in Mexican female carriers with hypohidrotic ectodermal dysplasia


Por: García-Delgado C., Noriega-Juárez M., Cervantes A., Abad-Flores J., Toledo-Bahena M., Valencia-Herrera A., Mena-Cedillos C., Villaseñor-Domínguez A., Sánchez-Boiso A., Akaki-Carreño Y., Río-Navarro B., Aguirre-Hernández J., López-López M., Cerbón M., Morán-Barroso V., Monroy-Jaramillo N.

Publicada: 1 ene 2020
Categoría: Dermatology

Resumen:
X-linked hypohidrotic ectodermal dysplasia (XLHED) is caused by EDA pathogenic variants. Female carriers show several clinical manifestations in variable percentages. We studied 11 Mexican heterozygous females with an EDA variant. The most frequent symptoms were similar to previous reports; however, two females (18%) reported dry eye syndrome, data rarely explored in carriers. The penetrance was 91% with clinical variability that might be related to the location of the mutation and/or to a skewed X-inactivation pattern. Our results highlight the importance of offering molecular testing to potential female carriers and support XLHED as an X-linked entity with incomplete penetrance in females. © 2020 Dermatologica Sinica Published by Wolters Kluwer - Medknow.

Filiaciones:
García-Delgado C.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Noriega-Juárez M.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Cervantes A.:
 Service of Genetics General Hospital of Mexico Dr. Eduardo Liceaga, Faculty of Medicine, National Autonomous University of Mexico, Mexico City, Mexico

Abad-Flores J.:
 Department of Genetics, National Institute of Neurology and Neurosurgery Manuel Velasco Suarez, Mexico City, Mexico

 Human Reproduction Research Unit, National Institute of Perinatology Isidro Espinosa de Los Reyes, Faculty of Chemistry, National Autonomous University of Mexico, Mexico City, Mexico

Toledo-Bahena M.:
 Department of Dermatology, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Valencia-Herrera A.:
 Department of Dermatology, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Mena-Cedillos C.:
 Department of Dermatology, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Villaseñor-Domínguez A.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Sánchez-Boiso A.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Akaki-Carreño Y.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

 Department of Dermatology, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Río-Navarro B.:
 Department of Immunology and Allergy, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Aguirre-Hernández J.:
 Laboratory of Genomics Genetics and Bioinformatics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

López-López M.:
 Department of Biological Systems, Autonomous Metropolitan University-Xochimilco, Mexico City, Mexico

Cerbón M.:
 Human Reproduction Research Unit, National Institute of Perinatology Isidro Espinosa de Los Reyes, Faculty of Chemistry, National Autonomous University of Mexico, Mexico City, Mexico

Morán-Barroso V.:
 Department of Genetics, Children's Hospital of Mexico Federico Gomez, Mexico City, Mexico

Monroy-Jaramillo N.:
 Department of Genetics, National Institute of Neurology and Neurosurgery Manuel Velasco Suarez, Mexico City, Mexico
ISSN: 10278117
Editorial
Elsevier Ltd, WOLTERS KLUWER INDIA PVT LTD , A-202, 2ND FLR, QUBE, C T S NO 1498A-2 VILLAGE MAROL, ANDHERI EAST, MUMBAI, 400059, INDIA, Taiwan
Tipo de documento: Article
Volumen: 38 Número: 3
Páginas: 172-175
WOS Id: 000601214900006