Literature DB >> 21164268

Adrenoleukodystrophy.

Marco Cappa, Carla Bizzarri, Catello Vollono, Anna Petroni, Sebastiano Banni.   

Abstract

X-linked adrenoleukodystrophy (ALD) is caused by mutations in the ABCD1 gene that encodes a protein of the peroxisomal membrane named ALDP. Mutations in ALDP result in elevated levels of very long chain fatty acids (VLCFA) and reduced VLCFA oxidation in peroxisomes. Three main phenotypes are seen in affected males. The childhood cerebral form manifests usually between ages 4 and 8 years. It initially resembles attention deficit disorder or hyperactivity. Progressive central demyelination with impairment of cognition, behavior, vision, hearing, and motor function follow the initial symptoms and often lead to total disability within 2 years. The second phenotype, adrenomyeloneuropathy, manifests most commonly in the late twenties as progressive paraparesis, sphincter disturbances, sexual dysfunction, and often, impaired adrenocortical function; all symptoms are progressive over decades. The third phenotype, 'Addison disease only', presents with primary adrenocortical insufficiency between age 2 years and adulthood and most commonly by age 7.5 years, without evidence of neurologic abnormality. Approximately 50% of females who are carriers develop neurologic manifestations that resemble adrenomyeloneuropathy but have a later onset (age ≥35 years) and a milder disease. In this review, we will give an overview of the present understanding of ALD, and the implications of new diagnostics and treatment.
Copyright © 2011 S. Karger AG, Basel.

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Year:  2010        PMID: 21164268     DOI: 10.1159/000321236

Source DB:  PubMed          Journal:  Endocr Dev        ISSN: 1421-7082


  8 in total

Review 1.  Transporters at CNS barrier sites: obstacles or opportunities for drug delivery?

Authors:  Lucy Sanchez-Covarrubias; Lauren M Slosky; Brandon J Thompson; Thomas P Davis; Patrick T Ronaldson
Journal:  Curr Pharm Des       Date:  2014       Impact factor: 3.116

2.  Partial rescue of Rett syndrome by ω-3 polyunsaturated fatty acids (PUFAs) oil.

Authors:  Claudio De Felice; Cinzia Signorini; Thierry Durand; Lucia Ciccoli; Silvia Leoncini; Maurizio D'Esposito; Stefania Filosa; Camille Oger; Alexandre Guy; Valérie Bultel-Poncé; Jean-Marie Galano; Alessandra Pecorelli; Laura De Felice; Giuseppe Valacchi; Joussef Hayek
Journal:  Genes Nutr       Date:  2012-03-08       Impact factor: 5.523

3.  F2-dihomo-isoprostanes as potential early biomarkers of lipid oxidative damage in Rett syndrome.

Authors:  Claudio De Felice; Cinzia Signorini; Thierry Durand; Camille Oger; Alexandre Guy; Valérie Bultel-Poncé; Jean-Marie Galano; Lucia Ciccoli; Silvia Leoncini; Maurizio D'Esposito; Stefania Filosa; Alessandra Pecorelli; Giuseppe Valacchi; Joussef Hayek
Journal:  J Lipid Res       Date:  2011-09-13       Impact factor: 5.922

4.  The absence of ABCD2 sensitizes mice to disruptions in lipid metabolism by dietary erucic acid.

Authors:  Jingjing Liu; Shuang Liang; Xiaoxi Liu; J Andrew Brown; Kylie E Newman; Manjula Sunkara; Andrew J Morris; Saloni Bhatnagar; Xiangan Li; Aurora Pujol; Gregory A Graf
Journal:  J Lipid Res       Date:  2012-04-06       Impact factor: 5.922

5.  Adrenoleukodystrophy: a forgotten diagnosis in children with primary Addison's disease.

Authors:  Marta Nascimento; Nádia Rodrigues; Filipa Espada; Marcelo Fonseca
Journal:  BMJ Case Rep       Date:  2012-08-21

6.  Exome sequencing identifies mutations in ABCD1 and DACH2 in two brothers with a distinct phenotype.

Authors:  Yanliang Zhang; Yanhui Liu; Ya Li; Yong Duan; Keyun Zhang; Junwang Wang; Yong Dai
Journal:  BMC Med Genet       Date:  2014-09-19       Impact factor: 2.103

7.  X-linked adult-onset adrenoleukodystrophy: Psychiatric and neurological manifestations.

Authors:  Daniah Shamim; Karen Alleyne
Journal:  SAGE Open Med Case Rep       Date:  2017-11-21

8.  Optic nerve demyelination as the presenting feature of adrenoleukodystrophy in a child.

Authors:  Nirupama Kasturi; Sandip Sarkar; Tanmay Gokhale; Chinnaiah G Delhikumar; Midhusha R Vendoti
Journal:  Indian J Ophthalmol       Date:  2022-07       Impact factor: 2.969

  8 in total

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