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A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene

Pseudohypoaldosteronism type 1 (PHA1) is a rare form of mineralocorticoid resistance characterized in newborns by salt wasting with dehydration, hyperkalemia and failure to thrive. This disease is heterogeneous in etiology and includes autosomal dominant PHA1 owing to mutations of the NR3C2 gene enc...

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Autores principales: Lee, Se Eun, Jung, Yun Hye, Han, Kyoung Hee, Lee, Hyun Kyung, Kang, Hee Gyung, Ha, Il Soo, Choi, Yong, Cheong, Hae Il
Formato: Texto
Lenguaje:English
Publicado: The Korean Pediatric Society 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3077507/
https://www.ncbi.nlm.nih.gov/pubmed/21503203
http://dx.doi.org/10.3345/kjp.2011.54.2.90
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author Lee, Se Eun
Jung, Yun Hye
Han, Kyoung Hee
Lee, Hyun Kyung
Kang, Hee Gyung
Ha, Il Soo
Choi, Yong
Cheong, Hae Il
author_facet Lee, Se Eun
Jung, Yun Hye
Han, Kyoung Hee
Lee, Hyun Kyung
Kang, Hee Gyung
Ha, Il Soo
Choi, Yong
Cheong, Hae Il
author_sort Lee, Se Eun
collection PubMed
description Pseudohypoaldosteronism type 1 (PHA1) is a rare form of mineralocorticoid resistance characterized in newborns by salt wasting with dehydration, hyperkalemia and failure to thrive. This disease is heterogeneous in etiology and includes autosomal dominant PHA1 owing to mutations of the NR3C2 gene encoding the mineralocorticoid receptor, autosomal recessive PHA1 due to mutations of the epithelial sodium channel (ENaC) gene, and secondary PHA1 associated with urinary tract diseases. Amongst these diseases, autosomal dominant PHA1 shows has manifestations restricted to renal tubules including a mild salt loss during infancy and that shows a gradual improvement with advancing age. Here, we report a neonatal case of PHA1 with a NR3C2 gene mutation (a heterozygous c.2146_2147insG in exon 5), in which the patient showed failure to thrive, hyponatremia, hyperkalemia, and elevated plasma renin and aldosterone levels. This is the first case of pseudohypoaldosteronism type 1 confirmed by genetic analysis in Korea.
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spelling pubmed-30775072011-04-18 A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene Lee, Se Eun Jung, Yun Hye Han, Kyoung Hee Lee, Hyun Kyung Kang, Hee Gyung Ha, Il Soo Choi, Yong Cheong, Hae Il Korean J Pediatr Case Report Pseudohypoaldosteronism type 1 (PHA1) is a rare form of mineralocorticoid resistance characterized in newborns by salt wasting with dehydration, hyperkalemia and failure to thrive. This disease is heterogeneous in etiology and includes autosomal dominant PHA1 owing to mutations of the NR3C2 gene encoding the mineralocorticoid receptor, autosomal recessive PHA1 due to mutations of the epithelial sodium channel (ENaC) gene, and secondary PHA1 associated with urinary tract diseases. Amongst these diseases, autosomal dominant PHA1 shows has manifestations restricted to renal tubules including a mild salt loss during infancy and that shows a gradual improvement with advancing age. Here, we report a neonatal case of PHA1 with a NR3C2 gene mutation (a heterozygous c.2146_2147insG in exon 5), in which the patient showed failure to thrive, hyponatremia, hyperkalemia, and elevated plasma renin and aldosterone levels. This is the first case of pseudohypoaldosteronism type 1 confirmed by genetic analysis in Korea. The Korean Pediatric Society 2011-02 2011-02-28 /pmc/articles/PMC3077507/ /pubmed/21503203 http://dx.doi.org/10.3345/kjp.2011.54.2.90 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
Lee, Se Eun
Jung, Yun Hye
Han, Kyoung Hee
Lee, Hyun Kyung
Kang, Hee Gyung
Ha, Il Soo
Choi, Yong
Cheong, Hae Il
A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title_full A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title_fullStr A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title_full_unstemmed A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title_short A case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
title_sort case of pseudohypoaldosteronism type 1 with a mutation in the mineralocorticoid receptor gene
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3077507/
https://www.ncbi.nlm.nih.gov/pubmed/21503203
http://dx.doi.org/10.3345/kjp.2011.54.2.90
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