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PMID: 30240710 Published · ppublish English Case Reports Journal Article

Autopsy findings of ectodermal dysplasia and sex development disorder in a fetus with 19q12q13 microdeletion.

European journal of medical genetics ·Vol. 62 ·No. 9 ·2019-09-00 ·页码 103539

Mottet N, Cabrol C, Metz JP, Toubin C, Arbez-Gindre F, Valduga M, McElreavey K, Riethmuller D, Van Maldergem L, Piard J

Abstract

A 5,6 Mb de novo 19q12-q13.12 interstitial deletion was diagnosed prenatally by array-comparative genomic hybridization in a 26 weeks male fetus presenting with intra-uterine growth retardation, left clubfoot, atypical genitalia and dysmorphic features. Autopsic examination following termination of pregnancy identified a severe disorder of sex development (DSD) including hypospadias, micropenis, bifid scrotum and right cryptorchidism associated with signs of ectodermal dysplasia: scalp hypopigmentation, thick and frizzy hair, absence of eyelashes, poorly developed nails and a thin skin with prominent superficial veins. Other findings were abnormal lung lobation and facial dysmorphism. This new case of DSD with a 19q12q13 deletion expands the phenotypic spectrum associated with this chromosomal rearrangment and suggests that WTIP is a strong candidate gene involved in male sex differentiation.

Keywords
19q12q13 deletion Disorder of sex development Ectodermal dysplasia Lung lobation
MeSH 主题词
Adult Chromosome Disorders/genetics,pathology Chromosomes, Human, Pair 19/genetics Co-Repressor Proteins/genetics Cytoskeletal Proteins/genetics Disorders of Sex Development/genetics,pathology Ectodermal Dysplasia/genetics,pathology Female Fetal Growth Retardation/genetics,pathology Gene Deletion Humans Male Pregnancy
化学物质
Co-Repressor Proteins Cytoskeletal Proteins WTIP protein, human
作者与单位
共 10 位作者,点击展开单位 / ORCID
Mottet Nicolas
Pole Mère-femme, Service de gynécologie Obstétrique, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France. Electronic address: n1mottet@chu-besancon.fr.
Cabrol Christelle
Centre de Génétique Humaine, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Metz Jean-Patrick
Pole Mère-femme, Service de gynécologie Obstétrique, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Toubin Claire
Pole Mère-femme, Service de gynécologie Obstétrique, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Arbez-Gindre Francine
Service d'Anatomie Pathologie, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Valduga Mylène
Laboratoire de génétique, Université de Nancy, France.
McElreavey Kenneth
Département de Biologie du Développement et cellules Souches, CNRS UMR 3738, Institut Pasteur, Paris, France.
Riethmuller Didier
Pole Mère-femme, Service de gynécologie Obstétrique, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Van Maldergem Lionel
Centre de Génétique Humaine, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Piard Juliette
Centre de Génétique Humaine, Centre Hospitalier Universitaire, Université de Franche-Comté, Besançon, France.
Article Info
Journal
European journal of medical genetics
Abbr.
Eur J Med Genet
ISSN
1878-0849
Corresponding email
Published
2019-09-00
电子出版
2018-00-18
页码
103539
Language
English
Country/Region
Netherlands
NLM ID
101247089
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