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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...
Autores principales: | , , , , , , , |
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Formato: | Texto |
Lenguaje: | English |
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The Korean Pediatric Society
2011
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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. |
format | Text |
id | pubmed-3077507 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2011 |
publisher | The Korean Pediatric Society |
record_format | MEDLINE/PubMed |
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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