Literature DB >> 33415088

Congenital Adrenal Hyperplasias Presenting in the Newborn and Young Infant.

Antonio Balsamo1, Federico Baronio1, Rita Ortolano1, Soara Menabo2, Lilia Baldazzi2, Valeria Di Natale1, Sofia Vissani1, Alessandra Cassio1.   

Abstract

Congenital adrenal hyperplasia includes autosomal recessive conditions that affect the adrenal cortex steroidogenic enzymes (cholesterol side-chain cleavage enzyme; 3β-hydroxysteroid dehydrogenase; 17α-hydroxylase/17,20 lyase; P450 oxidoreductase; 21-hydroxylase; and 11β-hydroxylase) and proteins (steroidogenic acute regulatory protein). These are located within the three major pathways of the steroidogenic apparatus involved in the production of mineralocorticoids, glucocorticoids, and androgens. Many countries have introduced newborn screening program (NSP) based on 17-OH-progesterone (17-OHP) immunoassays on dried blood spots, which enable faster diagnosis and treatment of the most severe forms of 21-hydroxylase deficiency (21-OHD). However, in several others, the use of this diagnostic tool has not yet been implemented and clinical diagnosis remains challenging, especially for males. Furthermore, less severe classic forms of 21-OHD and other rarer types of CAHs are not identified by NSP. The aim of this mini review is to highlight both the main clinical characteristics and therapeutic options of these conditions, which may be useful for a differential diagnosis in the neonatal period, while contributing to the biochemical evolution taking place in the steroidogenic field. Currently, chromatographic techniques coupled with tandem mass spectrometry are gaining attention due to an increase in the reliability of the test results of NPS for detecting 21-OHD. Furthermore, the possibility of identifying CAH patients that are not affected by 21-OHD but presenting elevated levels of 17-OHP by NSP and the opportunity to include the recently investigated 11-oxygenated androgens in the steroid profiles are promising tools for a more precise diagnosis and monitoring of some of these conditions.
Copyright © 2020 Balsamo, Baronio, Ortolano, Menabo, Baldazzi, Di Natale, Vissani and Cassio.

Entities:  

Keywords:  11-hydroxylase deficiency; 17-hydroxylase/17-20 lyase deficiency; 20–22-desmolase deficiency; 21-hydroxylase deficiency; 3-beta hydroxysteroid dehydrogenase deficiency; P-450 oxydoreductase deficiency; StAR deficiency; newborn

Year:  2020        PMID: 33415088      PMCID: PMC7783414          DOI: 10.3389/fped.2020.593315

Source DB:  PubMed          Journal:  Front Pediatr        ISSN: 2296-2360            Impact factor:   3.418


  81 in total

1.  High 17-hydroxyprogesterone level in newborn screening test for congenital adrenal hyperplasia.

Authors:  Yael Levy-Shraga; Orit Pinhas-Hamiel
Journal:  BMJ Case Rep       Date:  2016-02-24

Review 2.  MECHANISMS IN ENDOCRINOLOGY: Rare defects in adrenal steroidogenesis.

Authors:  Walter L Miller
Journal:  Eur J Endocrinol       Date:  2018-06-07       Impact factor: 6.664

3.  Restoration of mutant cytochrome P450 reductase activity by external flavin.

Authors:  Catherine Nicolo; Christa E Flück; Primus E Mullis; Amit V Pandey
Journal:  Mol Cell Endocrinol       Date:  2010-02-25       Impact factor: 4.102

4.  The role of a clinical score in the assessment of ambiguous genitalia.

Authors:  S F Ahmed; O Khwaja; I A Hughes
Journal:  BJU Int       Date:  2000-01       Impact factor: 5.588

5.  CYP11B1 mutations causing congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency.

Authors:  S Geley; K Kapelari; K Jöhrer; M Peter; J Glatzl; H Vierhapper; S Schwarz; A Helmberg; W G Sippell; P C White; R Kofler
Journal:  J Clin Endocrinol Metab       Date:  1996-08       Impact factor: 5.958

6.  Shaping parents, shaping penises: How medical teams frame parents' decisions in response to hypospadias.

Authors:  Katrina Roen; Peter Hegarty
Journal:  Br J Health Psychol       Date:  2018-07-27

7.  Characterizing Early Psychosocial Functioning of Parents of Children with Moderate to Severe Genital Ambiguity due to Disorders of Sex Development.

Authors:  Kristina I Suorsa; Alexandria J Mullins; Alayna P Tackett; Kristy J Scott Reyes; Paul Austin; Laurence Baskin; Kerlly Bernabé; Earl Cheng; Allyson Fried; Dominic Frimberger; Denise Galan; Lynette Gonzalez; Saul Greenfield; Bradley Kropp; Sabrina Meyer; Theresa Meyer; Natalie Nokoff; Blake Palmer; Dix Poppas; Alethea Paradis; Elizabeth Yerkes; Amy B Wisniewski; Larry L Mullins
Journal:  J Urol       Date:  2015-07-18       Impact factor: 7.450

8.  A novel mutation L260P of the steroidogenic acute regulatory protein gene in three unrelated patients of Swiss ancestry with congenital lipoid adrenal hyperplasia.

Authors:  Christa E Flück; Alexander Maret; Delphine Mallet; Stéphanie Portrat-Doyen; John C Achermann; Bruno Leheup; Gérald E Theintz; Primus E Mullis; Yves Morel
Journal:  J Clin Endocrinol Metab       Date:  2005-06-28       Impact factor: 5.958

9.  The pathophysiology and genetics of congenital lipoid adrenal hyperplasia.

Authors:  H S Bose; T Sugawara; J F Strauss; W L Miller
Journal:  N Engl J Med       Date:  1996-12-19       Impact factor: 91.245

10.  Heterozygous mutations in the cholesterol side-chain cleavage enzyme gene (CYP11A1) can cause transient adrenal insufficiency and life-threatening failure to thrive.

Authors:  Dimitrios T Papadimitriou; Christina Bothou; Diagoras Zarganis; Maria Karantza; Anastasios Papadimitriou
Journal:  Hormones (Athens)       Date:  2018-07-11       Impact factor: 2.885

View more
  4 in total

1.  A Novel Heterozygous Mutation of the CYP17A1 Gene in a Child with a Micropenis and Isolated 17,20-Lyase Deficiency.

Authors:  Maria Alessandra Saltarelli; Rossella Ferrante; Francesca Di Marcello; Daniela David; Silvia Valentinuzzi; Lucrezia Pilenzi; Luca Federici; Claudia Rossi; Liborio Stuppia; Stefano Tumini
Journal:  Int J Environ Res Public Health       Date:  2022-06-04       Impact factor: 4.614

2.  Exploring Dried Blood Spot Cortisol Concentrations as an Alternative for Monitoring Pediatric Adrenal Insufficiency Patients: A Model-Based Analysis.

Authors:  Viktoria Stachanow; Uta Neumann; Oliver Blankenstein; Davide Bindellini; Johanna Melin; Richard Ross; Martin J Whitaker; Wilhelm Huisinga; Robin Michelet; Charlotte Kloft
Journal:  Front Pharmacol       Date:  2022-03-17       Impact factor: 5.810

Review 3.  Adrenal hyperplasias in childhood: An update.

Authors:  Georgia Pitsava; Constantine A Stratakis
Journal:  Front Endocrinol (Lausanne)       Date:  2022-08-03       Impact factor: 6.055

Review 4.  Pediatric Adrenal Insufficiency: Challenges and Solutions.

Authors:  Daniela Nisticò; Benedetta Bossini; Simone Benvenuto; Maria Chiara Pellegrin; Gianluca Tornese
Journal:  Ther Clin Risk Manag       Date:  2022-01-11       Impact factor: 2.423

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.