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A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay

Cleft lip and/or cleft palate are a common group of birth defects that further classify into syndromic and non-syndromic forms. The syndromic forms are usually accompanied by additional physical or cognitive abnormalities. Isolated cleft palate syndromes are less common; however, they are associated...

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Autores principales: Hexner-Erlichman, Zufit, Fichtman, Boris, Zehavi, Yoav, Khayat, Morad, Jabaly-Habib, Haneen, Izhaki-Tavor, Lee S., Dessau, Moshe, Elpeleg, Orly, Spiegel, Ronen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9152136/
https://www.ncbi.nlm.nih.gov/pubmed/35656379
http://dx.doi.org/10.3389/fped.2022.859034
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author Hexner-Erlichman, Zufit
Fichtman, Boris
Zehavi, Yoav
Khayat, Morad
Jabaly-Habib, Haneen
Izhaki-Tavor, Lee S.
Dessau, Moshe
Elpeleg, Orly
Spiegel, Ronen
author_facet Hexner-Erlichman, Zufit
Fichtman, Boris
Zehavi, Yoav
Khayat, Morad
Jabaly-Habib, Haneen
Izhaki-Tavor, Lee S.
Dessau, Moshe
Elpeleg, Orly
Spiegel, Ronen
author_sort Hexner-Erlichman, Zufit
collection PubMed
description Cleft lip and/or cleft palate are a common group of birth defects that further classify into syndromic and non-syndromic forms. The syndromic forms are usually accompanied by additional physical or cognitive abnormalities. Isolated cleft palate syndromes are less common; however, they are associated with a variety of congenital malformations and generally have an underlying genetic etiology. A single report in 2019 described a novel syndrome in three individuals, characterized by cleft palate, developmental delay and proliferative retinopathy due to a homozygous non-sense mutation in the LRRC32 gene encoding glycoprotein A repetitions predominant (GARP), a cell surface polypeptide crucial for the processing and maturation of transforming growth factor β (TGF-β). We describe a patient who presented with cleft palate, prenatal and postnatal severe growth retardation, global developmental delay, dysmorphic facial features and progressive vitreoretinopathy. Whole exome sequencing (WES) revealed a very rare homozygous missense variant in the LRRC32 gene, which resulted in substitution of a highly conserved isoleucine to threonine. Protein modeling suggested this variant may negatively affect GARP function on latent TGF-β activation. In summary, our report further expands the clinical features of cleft palate, proliferative retinopathy and developmental delay syndrome and emphasizes the association of LRRC32 pathogenic variants with this new syndrome.
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spelling pubmed-91521362022-06-01 A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay Hexner-Erlichman, Zufit Fichtman, Boris Zehavi, Yoav Khayat, Morad Jabaly-Habib, Haneen Izhaki-Tavor, Lee S. Dessau, Moshe Elpeleg, Orly Spiegel, Ronen Front Pediatr Pediatrics Cleft lip and/or cleft palate are a common group of birth defects that further classify into syndromic and non-syndromic forms. The syndromic forms are usually accompanied by additional physical or cognitive abnormalities. Isolated cleft palate syndromes are less common; however, they are associated with a variety of congenital malformations and generally have an underlying genetic etiology. A single report in 2019 described a novel syndrome in three individuals, characterized by cleft palate, developmental delay and proliferative retinopathy due to a homozygous non-sense mutation in the LRRC32 gene encoding glycoprotein A repetitions predominant (GARP), a cell surface polypeptide crucial for the processing and maturation of transforming growth factor β (TGF-β). We describe a patient who presented with cleft palate, prenatal and postnatal severe growth retardation, global developmental delay, dysmorphic facial features and progressive vitreoretinopathy. Whole exome sequencing (WES) revealed a very rare homozygous missense variant in the LRRC32 gene, which resulted in substitution of a highly conserved isoleucine to threonine. Protein modeling suggested this variant may negatively affect GARP function on latent TGF-β activation. In summary, our report further expands the clinical features of cleft palate, proliferative retinopathy and developmental delay syndrome and emphasizes the association of LRRC32 pathogenic variants with this new syndrome. Frontiers Media S.A. 2022-05-17 /pmc/articles/PMC9152136/ /pubmed/35656379 http://dx.doi.org/10.3389/fped.2022.859034 Text en Copyright © 2022 Hexner-Erlichman, Fichtman, Zehavi, Khayat, Jabaly-Habib, Izhaki-Tavor, Dessau, Elpeleg and Spiegel. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Hexner-Erlichman, Zufit
Fichtman, Boris
Zehavi, Yoav
Khayat, Morad
Jabaly-Habib, Haneen
Izhaki-Tavor, Lee S.
Dessau, Moshe
Elpeleg, Orly
Spiegel, Ronen
A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title_full A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title_fullStr A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title_full_unstemmed A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title_short A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay
title_sort novel homozygous missense variant in the lrrc32 gene is associated with a new syndrome of cleft palate, progressive vitreoretinopathy, growth retardation, and developmental delay
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9152136/
https://www.ncbi.nlm.nih.gov/pubmed/35656379
http://dx.doi.org/10.3389/fped.2022.859034
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