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Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations

Primary hypokalemic periodic paralysis (HOKPP) is an autosomal dominant disorder manifesting as recurrent periodic flaccid paralysis and concomitant hypokalemia. HOKPP is divided into type 1 and type 2 based on the causative gene. Although 2 different ion channels have been identified as the molecul...

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Autores principales: Kim, Hunmin, Hwang, Hee, Cheong, Hae Il, Park, Hye Won
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Korean Pediatric Society 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3254894/
https://www.ncbi.nlm.nih.gov/pubmed/22253645
http://dx.doi.org/10.3345/kjp.2011.54.11.473
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author Kim, Hunmin
Hwang, Hee
Cheong, Hae Il
Park, Hye Won
author_facet Kim, Hunmin
Hwang, Hee
Cheong, Hae Il
Park, Hye Won
author_sort Kim, Hunmin
collection PubMed
description Primary hypokalemic periodic paralysis (HOKPP) is an autosomal dominant disorder manifesting as recurrent periodic flaccid paralysis and concomitant hypokalemia. HOKPP is divided into type 1 and type 2 based on the causative gene. Although 2 different ion channels have been identified as the molecular genetic cause of HOKPP, the clinical manifestations between the 2 groups are similar. We report the cases of 2 patients with HOKPP who both presented with typical clinical manifestations, but with mutations in 2 different genes (CACNA1Sp.Arg528His and SCN4A p.Arg672His). Despite the similar clinical manifestations, there were differences in the response to acetazolamide treatment between certain genotypes of SCN4A mutations and CACNA1S mutations. We identified p.Arg672His in the SCN4A gene of patient 2 immediately after the first attack through a molecular genetic testing strategy. Molecular genetic diagnosis is important for genetic counseling and selecting preventive treatment.
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spelling pubmed-32548942012-01-17 Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations Kim, Hunmin Hwang, Hee Cheong, Hae Il Park, Hye Won Korean J Pediatr Case Report Primary hypokalemic periodic paralysis (HOKPP) is an autosomal dominant disorder manifesting as recurrent periodic flaccid paralysis and concomitant hypokalemia. HOKPP is divided into type 1 and type 2 based on the causative gene. Although 2 different ion channels have been identified as the molecular genetic cause of HOKPP, the clinical manifestations between the 2 groups are similar. We report the cases of 2 patients with HOKPP who both presented with typical clinical manifestations, but with mutations in 2 different genes (CACNA1Sp.Arg528His and SCN4A p.Arg672His). Despite the similar clinical manifestations, there were differences in the response to acetazolamide treatment between certain genotypes of SCN4A mutations and CACNA1S mutations. We identified p.Arg672His in the SCN4A gene of patient 2 immediately after the first attack through a molecular genetic testing strategy. Molecular genetic diagnosis is important for genetic counseling and selecting preventive treatment. The Korean Pediatric Society 2011-11 2011-11-30 /pmc/articles/PMC3254894/ /pubmed/22253645 http://dx.doi.org/10.3345/kjp.2011.54.11.473 Text en Copyright © 2011 by The Korean Pediatric Society http://creativecommons.org/licenses/by-nc/3.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Kim, Hunmin
Hwang, Hee
Cheong, Hae Il
Park, Hye Won
Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title_full Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title_fullStr Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title_full_unstemmed Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title_short Hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
title_sort hypokalemic periodic paralysis; two different genes responsible for similar clinical manifestations
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3254894/
https://www.ncbi.nlm.nih.gov/pubmed/22253645
http://dx.doi.org/10.3345/kjp.2011.54.11.473
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