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Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins

Background and objectives: Coffin-Lowry Syndrome (CLS), a rare neurodegenerative disorder, is mainly diagnosed based on clinical manifestations and molecular analyses. In total, about 20 cases of CLS have been reported in China. Here, we report two cases of CLS in identical twin brothers and examine...

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Autores principales: Jin, Huiying, Li, Haifeng, Qiang, Shu
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9320784/
https://www.ncbi.nlm.nih.gov/pubmed/35888677
http://dx.doi.org/10.3390/medicina58070958
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author Jin, Huiying
Li, Haifeng
Qiang, Shu
author_facet Jin, Huiying
Li, Haifeng
Qiang, Shu
author_sort Jin, Huiying
collection PubMed
description Background and objectives: Coffin-Lowry Syndrome (CLS), a rare neurodegenerative disorder, is mainly diagnosed based on clinical manifestations and molecular analyses. In total, about 20 cases of CLS have been reported in China. Here, we report two cases of CLS in identical twin brothers and examine their potential causative mutations. Methods: The Trio mode was used in this analysis, i.e., DNA from the proband and his parents was sequenced. Furthermore, DNA from the proband’s twin brother was used for confirmation. Results: A hemizygous variation was detected in the 11th exon of the RPS6KA3 gene, c.898C>T (p.R300*) of the proband, and the same site variation was detected in his identical twin brother; however, the mutation was not detected in his parents. Conclusions: The RPS6KA3 gene mutation c.898C>T (p.R300*) is the causative factor of familial CLS. The variant detected was reported for the first time in the Chinese population. Additionally, by analyzing the previous literature, we were able to summarize the phenotypic and genetic characteristics of GLS in China.
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spelling pubmed-93207842022-07-27 Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins Jin, Huiying Li, Haifeng Qiang, Shu Medicina (Kaunas) Case Report Background and objectives: Coffin-Lowry Syndrome (CLS), a rare neurodegenerative disorder, is mainly diagnosed based on clinical manifestations and molecular analyses. In total, about 20 cases of CLS have been reported in China. Here, we report two cases of CLS in identical twin brothers and examine their potential causative mutations. Methods: The Trio mode was used in this analysis, i.e., DNA from the proband and his parents was sequenced. Furthermore, DNA from the proband’s twin brother was used for confirmation. Results: A hemizygous variation was detected in the 11th exon of the RPS6KA3 gene, c.898C>T (p.R300*) of the proband, and the same site variation was detected in his identical twin brother; however, the mutation was not detected in his parents. Conclusions: The RPS6KA3 gene mutation c.898C>T (p.R300*) is the causative factor of familial CLS. The variant detected was reported for the first time in the Chinese population. Additionally, by analyzing the previous literature, we were able to summarize the phenotypic and genetic characteristics of GLS in China. MDPI 2022-07-20 /pmc/articles/PMC9320784/ /pubmed/35888677 http://dx.doi.org/10.3390/medicina58070958 Text en © 2022 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 (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Jin, Huiying
Li, Haifeng
Qiang, Shu
Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title_full Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title_fullStr Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title_full_unstemmed Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title_short Coffin-Lowry Syndrome Induced by RPS6KA3 Gene Variation in China: A Case Report in Twins
title_sort coffin-lowry syndrome induced by rps6ka3 gene variation in china: a case report in twins
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9320784/
https://www.ncbi.nlm.nih.gov/pubmed/35888677
http://dx.doi.org/10.3390/medicina58070958
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