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Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome

Mowat-Wilson syndrome is a rare neurodevelopmental disorder caused by pathogenic variants in the ZEB2 gene, intragenic deletions of the ZEB2 gene, and microdeletions in the critical chromosomal region 2q22-23, where the ZEB2 gene is located. Mowat-Wilson syndrome is characterized by typical facial f...

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Autores principales: Jakubiak, Aleksandra, Szczałuba, Krzysztof, Badura-Stronka, Magdalena, Kutkowska-Kaźmierczak, Anna, Jakubiuk-Tomaszuk, Anna, Chilarska, Tatiana, Pilch, Jacek, Braun-Walicka, Natalia, Castaneda, Jennifer, Wołyńska, Katarzyna, Wiśniewska, Marzena, Kugaudo, Monika, Bielecka, Monika, Pesz, Karolina, Wierzba, Jolanta, Latos-Bieleńska, Anna, Obersztyn, Ewa, Krajewska-Walasek, Małgorzata, Śmigiel, Robert
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8357696/
https://www.ncbi.nlm.nih.gov/pubmed/33982229
http://dx.doi.org/10.1007/s13353-021-00636-1
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author Jakubiak, Aleksandra
Szczałuba, Krzysztof
Badura-Stronka, Magdalena
Kutkowska-Kaźmierczak, Anna
Jakubiuk-Tomaszuk, Anna
Chilarska, Tatiana
Pilch, Jacek
Braun-Walicka, Natalia
Castaneda, Jennifer
Wołyńska, Katarzyna
Wiśniewska, Marzena
Kugaudo, Monika
Bielecka, Monika
Pesz, Karolina
Wierzba, Jolanta
Latos-Bieleńska, Anna
Obersztyn, Ewa
Krajewska-Walasek, Małgorzata
Śmigiel, Robert
author_facet Jakubiak, Aleksandra
Szczałuba, Krzysztof
Badura-Stronka, Magdalena
Kutkowska-Kaźmierczak, Anna
Jakubiuk-Tomaszuk, Anna
Chilarska, Tatiana
Pilch, Jacek
Braun-Walicka, Natalia
Castaneda, Jennifer
Wołyńska, Katarzyna
Wiśniewska, Marzena
Kugaudo, Monika
Bielecka, Monika
Pesz, Karolina
Wierzba, Jolanta
Latos-Bieleńska, Anna
Obersztyn, Ewa
Krajewska-Walasek, Małgorzata
Śmigiel, Robert
author_sort Jakubiak, Aleksandra
collection PubMed
description Mowat-Wilson syndrome is a rare neurodevelopmental disorder caused by pathogenic variants in the ZEB2 gene, intragenic deletions of the ZEB2 gene, and microdeletions in the critical chromosomal region 2q22-23, where the ZEB2 gene is located. Mowat-Wilson syndrome is characterized by typical facial features that change with the age, severe developmental delay with intellectual disability, and multiple congenital abnormalities. The authors describe the clinical and genetic aspects of 28th patients with Mowat-Wilson syndrome diagnosed in Poland. Characteristic dysmorphic features, psychomotor retardation, intellectual disability, and congenital anomalies were present in all cases. The incidence of most common congenital anomalies (heart defect, Hirschsprung disease, brain defects) was similar to presented in literature. Epilepsy was less common compared to previously reported cases. Although the spectrum of disorders in patients with Mowat-Wilson syndrome is wide, knowledge of characteristic dysmorphic features awareness of accompanying abnormalities, especially intellectual disability, improves detection of the syndrome.
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spelling pubmed-83576962021-08-30 Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome Jakubiak, Aleksandra Szczałuba, Krzysztof Badura-Stronka, Magdalena Kutkowska-Kaźmierczak, Anna Jakubiuk-Tomaszuk, Anna Chilarska, Tatiana Pilch, Jacek Braun-Walicka, Natalia Castaneda, Jennifer Wołyńska, Katarzyna Wiśniewska, Marzena Kugaudo, Monika Bielecka, Monika Pesz, Karolina Wierzba, Jolanta Latos-Bieleńska, Anna Obersztyn, Ewa Krajewska-Walasek, Małgorzata Śmigiel, Robert J Appl Genet Human Genetics • Original Paper Mowat-Wilson syndrome is a rare neurodevelopmental disorder caused by pathogenic variants in the ZEB2 gene, intragenic deletions of the ZEB2 gene, and microdeletions in the critical chromosomal region 2q22-23, where the ZEB2 gene is located. Mowat-Wilson syndrome is characterized by typical facial features that change with the age, severe developmental delay with intellectual disability, and multiple congenital abnormalities. The authors describe the clinical and genetic aspects of 28th patients with Mowat-Wilson syndrome diagnosed in Poland. Characteristic dysmorphic features, psychomotor retardation, intellectual disability, and congenital anomalies were present in all cases. The incidence of most common congenital anomalies (heart defect, Hirschsprung disease, brain defects) was similar to presented in literature. Epilepsy was less common compared to previously reported cases. Although the spectrum of disorders in patients with Mowat-Wilson syndrome is wide, knowledge of characteristic dysmorphic features awareness of accompanying abnormalities, especially intellectual disability, improves detection of the syndrome. Springer Berlin Heidelberg 2021-05-12 2021 /pmc/articles/PMC8357696/ /pubmed/33982229 http://dx.doi.org/10.1007/s13353-021-00636-1 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Human Genetics • Original Paper
Jakubiak, Aleksandra
Szczałuba, Krzysztof
Badura-Stronka, Magdalena
Kutkowska-Kaźmierczak, Anna
Jakubiuk-Tomaszuk, Anna
Chilarska, Tatiana
Pilch, Jacek
Braun-Walicka, Natalia
Castaneda, Jennifer
Wołyńska, Katarzyna
Wiśniewska, Marzena
Kugaudo, Monika
Bielecka, Monika
Pesz, Karolina
Wierzba, Jolanta
Latos-Bieleńska, Anna
Obersztyn, Ewa
Krajewska-Walasek, Małgorzata
Śmigiel, Robert
Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title_full Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title_fullStr Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title_full_unstemmed Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title_short Clinical characteristics of Polish patients with molecularly confirmed Mowat-Wilson syndrome
title_sort clinical characteristics of polish patients with molecularly confirmed mowat-wilson syndrome
topic Human Genetics • Original Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8357696/
https://www.ncbi.nlm.nih.gov/pubmed/33982229
http://dx.doi.org/10.1007/s13353-021-00636-1
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