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Kabuki Syndrome—Clinical Review with Molecular Aspects

Kabuki syndrome (KS) is a rare developmental disorder principally comprised of developmental delay, hypotonia and a clearly defined dysmorphism: elongation of the structures surrounding the eyes, a shortened and depressed nose, thinning of the upper lip and thickening of the lower lip, large and pro...

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Autores principales: Boniel, Snir, Szymańska, Krystyna, Śmigiel, Robert, Szczałuba, Krzysztof
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8064399/
https://www.ncbi.nlm.nih.gov/pubmed/33805950
http://dx.doi.org/10.3390/genes12040468
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author Boniel, Snir
Szymańska, Krystyna
Śmigiel, Robert
Szczałuba, Krzysztof
author_facet Boniel, Snir
Szymańska, Krystyna
Śmigiel, Robert
Szczałuba, Krzysztof
author_sort Boniel, Snir
collection PubMed
description Kabuki syndrome (KS) is a rare developmental disorder principally comprised of developmental delay, hypotonia and a clearly defined dysmorphism: elongation of the structures surrounding the eyes, a shortened and depressed nose, thinning of the upper lip and thickening of the lower lip, large and prominent ears, hypertrichosis and scoliosis. Other characteristics include poor physical growth, cardiac, gastrointestinal and renal anomalies as well as variable behavioral issues, including autistic features. De novo or inherited pathogenic/likely pathogenic variants in the KMT2D gene are the most common cause of KS and account for up to 75% of patients. Variants in KDM6A cause up to 5% of cases (X-linked dominant inheritance), while the etiology of about 20% of cases remains unknown. Current KS diagnostic criteria include hypotonia during infancy, developmental delay and/or intellectual disability, typical dysmorphism and confirmed pathogenic/likely pathogenic variant in KMT2D or KDM6A. Care for KS patients includes the control of physical and psychomotor development during childhood, rehabilitation and multi-specialist care. This paper reviews the current clinical knowledge, provides molecular and scientific links and sheds light on the treatment of Kabuki syndrome individuals.
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spelling pubmed-80643992021-04-24 Kabuki Syndrome—Clinical Review with Molecular Aspects Boniel, Snir Szymańska, Krystyna Śmigiel, Robert Szczałuba, Krzysztof Genes (Basel) Review Kabuki syndrome (KS) is a rare developmental disorder principally comprised of developmental delay, hypotonia and a clearly defined dysmorphism: elongation of the structures surrounding the eyes, a shortened and depressed nose, thinning of the upper lip and thickening of the lower lip, large and prominent ears, hypertrichosis and scoliosis. Other characteristics include poor physical growth, cardiac, gastrointestinal and renal anomalies as well as variable behavioral issues, including autistic features. De novo or inherited pathogenic/likely pathogenic variants in the KMT2D gene are the most common cause of KS and account for up to 75% of patients. Variants in KDM6A cause up to 5% of cases (X-linked dominant inheritance), while the etiology of about 20% of cases remains unknown. Current KS diagnostic criteria include hypotonia during infancy, developmental delay and/or intellectual disability, typical dysmorphism and confirmed pathogenic/likely pathogenic variant in KMT2D or KDM6A. Care for KS patients includes the control of physical and psychomotor development during childhood, rehabilitation and multi-specialist care. This paper reviews the current clinical knowledge, provides molecular and scientific links and sheds light on the treatment of Kabuki syndrome individuals. MDPI 2021-03-25 /pmc/articles/PMC8064399/ /pubmed/33805950 http://dx.doi.org/10.3390/genes12040468 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) ).
spellingShingle Review
Boniel, Snir
Szymańska, Krystyna
Śmigiel, Robert
Szczałuba, Krzysztof
Kabuki Syndrome—Clinical Review with Molecular Aspects
title Kabuki Syndrome—Clinical Review with Molecular Aspects
title_full Kabuki Syndrome—Clinical Review with Molecular Aspects
title_fullStr Kabuki Syndrome—Clinical Review with Molecular Aspects
title_full_unstemmed Kabuki Syndrome—Clinical Review with Molecular Aspects
title_short Kabuki Syndrome—Clinical Review with Molecular Aspects
title_sort kabuki syndrome—clinical review with molecular aspects
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8064399/
https://www.ncbi.nlm.nih.gov/pubmed/33805950
http://dx.doi.org/10.3390/genes12040468
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