Literature DB >> 28593901

Pseudohypoaldosteronism types I and II: little more than a name in common.

Dídac Casas-Alba1, Jordi Vila Cots2, Laura Monfort Carretero1, Loreto Martorell Sampol3, Maria-Christina Zennaro4, Xavier Jeunemaitre4, Juan Antonio Camacho Díaz2.   

Abstract

Pseudohypoaldosteronism (PHA) comprises a diverse group of rare diseases characterized by sodium and potassium imbalances incorrectly attributed to a defect in aldosterone production. Two different forms of PHA have been described, type I (PHAI) and type II (PHAII). PHAI has been subclassified into renal and systemic. Given the rarity and heterogeneity of this group of disorders we report three patients who carry PHA and a brief revision of current literature focused on the comparative analysis of PHAI and PHAII. Cases 1 and 2 presented with hyponatremia, hyperkalemia, metabolic acidosis and elevated plasma aldosterone and plasma renin activity in the neonatal period. Sequence analysis of the NRC2 gene demonstrated a novel heterozygous c.403C>T mutation in case 1 and a complete deletion in case 2, confirming the diagnosis of renal PHAI. Case 3 was a 4-year-old with hypertension, hyperkalemia, metabolic acidosis, normal plasma aldosterone and decreased plasma renin activity. Sequence analysis of the CUL3 gene demonstrated a previously unreported heterozygous c.1377+2T>3 mutation, confirming the diagnosis of PHAII-E. We highlight the importance of the determination of plasma aldosterone and plasma renin activity in the context of persistent sodium and potassium imbalances in children.

Entities:  

Keywords:  CUL3 gene; NR3C2 gene; aldosterone; pseudohypoaldosteronism; renin activity

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Year:  2017        PMID: 28593901     DOI: 10.1515/jpem-2016-0467

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  4 in total

1.  An infant with hyponatremia, hyperkalemia, and metabolic acidosis associated with urinary tract infection: Answers.

Authors:  Bahriye Atmis; İhsan Turan; Engin Melek; Aysun Karabay Bayazit
Journal:  Pediatr Nephrol       Date:  2019-05-03       Impact factor: 3.714

2.  Interstitial 4q Deletion Syndrome Including NR3C2 Causing Pseudohypoaldosteronism.

Authors:  Amanda Barone Pritchard; Alyssa Ritter; Hutton M Kearney; Kosuke Izumi
Journal:  Mol Syndromol       Date:  2019-12-21

3.  When salt is needed to grow: Answers.

Authors:  Ester Conversano; Sara Romano; Andrea Taddio; Flavio Faletra; Davide Zanon; Egidio Barbi; Marco Pennesi
Journal:  Pediatr Nephrol       Date:  2020-08-10       Impact factor: 3.714

4.  De novo variants in CUL3 are associated with global developmental delays with or without infantile spasms.

Authors:  Mitsuko Nakashima; Mitsuhiro Kato; Masaru Matsukura; Ryutaro Kira; Lock-Hock Ngu; Klaske D Lichtenbelt; Koen L I van Gassen; Satomi Mitsuhashi; Hirotomo Saitsu; Naomichi Matsumoto
Journal:  J Hum Genet       Date:  2020-04-27       Impact factor: 3.172

  4 in total

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