Skewed X-Chromosome Inactivation in EDA-Related X-Linked Hypohidrotic Ectodermal Dysplasia in a Moroccan Family

X-linked hypohidrotic ectodermal dysplasia (HED) is a rare genetic disorder caused by pathogenic variants in the EDA gene and characterized by variable involvement of ectoderm-derived tissues. The aim of this study is to describe the clinical and molecular features of a Moroccan family affected by X-linked HED, with particular emphasis on the role of X-chromosome inactivation in phenotypic variability among female carriers. Clinical and genetic investigations identified the pathogenic EDA variant c.467G>A (p.Arg156His) in an affected boy and his heterozygous mother. Notably, the mother exhibited a more severe ectodermal phenotype than typically observed in female carriers. This observation highlights the likely impact of skewed X-chromosome inactivation on disease expression and underscores its importance for genotype–phenotype interpretation, genetic counseling, and multidisciplinary care.

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Publication Details

Journal
Zenodo (CERN European Organization for Nuclear Research)
Published
2026-02-23
DOI
https://doi.org/10.5281/zenodo.18766670
Primary Topic
dental development and anomalies
Type
article
Field-Weighted Citation Impact
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article

Skewed X-Chromosome Inactivation in EDA-Related X-Linked Hypohidrotic Ectodermal Dysplasia in a Moroccan Family

Fatima Maachi, Hind Dehbi, M. Hammouch, S. Chiheb et al.
Zenodo (CERN European Organization for Nuclear Research)
dental development and anomalies
article

Skewed X-Chromosome Inactivation in EDA-Related X-Linked Hypohidrotic Ectodermal Dysplasia in a Moroccan Family

Fatima Maachi, Hind Dehbi, M. Hammouch, S. Chiheb, F.Z. Elfatoik
article en

Abstract

X-linked hypohidrotic ectodermal dysplasia (HED) is a rare genetic disorder caused by pathogenic variants in the EDA gene and characterized by variable involvement of ectoderm-derived tissues. The aim of this study is to describe the clinical and molecular features of a Moroccan family affected by X-linked HED, with particular emphasis on the role of X-chromosome inactivation in phenotypic variability among female carriers. Clinical and genetic investigations identified the pathogenic EDA variant c.467G>A (p.Arg156His) in an affected boy and his heterozygous mother. Notably, the mother exhibited a more severe ectodermal phenotype than typically observed in female carriers. This observation highlights the likely impact of skewed X-chromosome inactivation on disease expression and underscores its importance for genotype–phenotype interpretation, genetic counseling, and multidisciplinary care.

Zenodo (CERN European Organization for Nuclear Research)
Centre Hospitalier Universitaire Ibn Rochd (MA), University of Hassan II Casablanca (MA)
Openalex Percentile: Top 43%
dental development and anomalies
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