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O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)

O'Donnel-Luria-Rodan (ODLURO) syndrome is a neurodevelopmental disorder with autosomal dominant inheritance. It appears more frequently in males during the first decade of life and is associated with developmental delay, low intelligence quotient, autism spectrum disorder-like behavior, epileps...

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Autores principales: Cătană, Andreea, Kutasi, Enikő, Cuzmici-Barabaș, Zina, Militaru, Diana, Iordănescu, Irina, Militaru, Mariela Sanda
Formato: Online Artículo Texto
Lenguaje:English
Publicado: D.A. Spandidos 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9016787/
https://www.ncbi.nlm.nih.gov/pubmed/35481221
http://dx.doi.org/10.3892/etm.2022.11294
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author Cătană, Andreea
Kutasi, Enikő
Cuzmici-Barabaș, Zina
Militaru, Diana
Iordănescu, Irina
Militaru, Mariela Sanda
author_facet Cătană, Andreea
Kutasi, Enikő
Cuzmici-Barabaș, Zina
Militaru, Diana
Iordănescu, Irina
Militaru, Mariela Sanda
author_sort Cătană, Andreea
collection PubMed
description O'Donnel-Luria-Rodan (ODLURO) syndrome is a neurodevelopmental disorder with autosomal dominant inheritance. It appears more frequently in males during the first decade of life and is associated with developmental delay, low intelligence quotient, autism spectrum disorder-like behavior, epilepsy, speech delay, aggression, facial and skeletal deformities, gastrointestinal symptoms and hypotonia. Although few cases have been documented, it appears that the phenotype spectrum may vary, especially between the two biological sexes. The present study reported a case of a 5-year-old male patient who was diagnosed with ODLURO at the age of 4 years using whole-exome sequencing. Molecular analysis identified a new mutation in the lysine methyltransferase 2E (inactive) (KMT2E) gene, which was classified as a variant with unknown significance. The father, who presented with non-specific and undiagnosed psychiatric manifestations, presented the same KMT2E variant. The case described in the present study is not only interesting because there are <40 cases described in the literature, but also because a new inherited mutation in the KMT2E gene, present in both father and son, that resulted in different phenotypic manifestations was identified.
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spelling pubmed-90167872022-04-26 O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report) Cătană, Andreea Kutasi, Enikő Cuzmici-Barabaș, Zina Militaru, Diana Iordănescu, Irina Militaru, Mariela Sanda Exp Ther Med Case Report O'Donnel-Luria-Rodan (ODLURO) syndrome is a neurodevelopmental disorder with autosomal dominant inheritance. It appears more frequently in males during the first decade of life and is associated with developmental delay, low intelligence quotient, autism spectrum disorder-like behavior, epilepsy, speech delay, aggression, facial and skeletal deformities, gastrointestinal symptoms and hypotonia. Although few cases have been documented, it appears that the phenotype spectrum may vary, especially between the two biological sexes. The present study reported a case of a 5-year-old male patient who was diagnosed with ODLURO at the age of 4 years using whole-exome sequencing. Molecular analysis identified a new mutation in the lysine methyltransferase 2E (inactive) (KMT2E) gene, which was classified as a variant with unknown significance. The father, who presented with non-specific and undiagnosed psychiatric manifestations, presented the same KMT2E variant. The case described in the present study is not only interesting because there are <40 cases described in the literature, but also because a new inherited mutation in the KMT2E gene, present in both father and son, that resulted in different phenotypic manifestations was identified. D.A. Spandidos 2022-05 2022-04-04 /pmc/articles/PMC9016787/ /pubmed/35481221 http://dx.doi.org/10.3892/etm.2022.11294 Text en Copyright: © Cătană et al. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License (https://creativecommons.org/licenses/by-nc-nd/4.0/) , which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made.
spellingShingle Case Report
Cătană, Andreea
Kutasi, Enikő
Cuzmici-Barabaș, Zina
Militaru, Diana
Iordănescu, Irina
Militaru, Mariela Sanda
O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title_full O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title_fullStr O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title_full_unstemmed O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title_short O'Donnel-Luria-Rodan Syndrome: New gene variant identified in Romania (A case report)
title_sort o'donnel-luria-rodan syndrome: new gene variant identified in romania (a case report)
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9016787/
https://www.ncbi.nlm.nih.gov/pubmed/35481221
http://dx.doi.org/10.3892/etm.2022.11294
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