Literature DB >> 3508688

Myopathy in an infant with a fatal peroxisomal disorder.

J Wolff1, W L Nyhan, H Powell, D Takahashi, J Hutzler, A K Hajra, N S Datta, I Singh, H W Moser.   

Abstract

An infant with neonatal adrenoleukodystrophy experienced extreme hypotonia and virtually continuous convulsions at four months of age and died. Light and electron microscopic examination revealed evidence of myopathy and the presence of mitochondrial inclusions. Concentrations of very long-chain fatty acids were elevated in blood and fibroblasts and the oxidation of 14C-labeled fatty acids was defective. Urinary pipecolic acid content was increased. Activity of the peroxisomal dihydroxyacetone phosphate acyltransferase, which catalyzes the first step in plasmalogen synthesis, was decreased.

Entities:  

Mesh:

Substances:

Year:  1986        PMID: 3508688     DOI: 10.1016/0887-8994(86)90004-4

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  8 in total

1.  A pathological study of a peripheral nerve in a case of neonatal adrenoleukodystrophy.

Authors:  T Mito; K Takada; S Akaboshi; S Takashima; K Takeshita; Y Origuchi
Journal:  Acta Neuropathol       Date:  1989       Impact factor: 17.088

Review 2.  Inherited peroxisomal disorders involving the nervous system.

Authors:  J B Stephenson
Journal:  Arch Dis Child       Date:  1988-07       Impact factor: 3.791

3.  Genotype-phenotype correlations and disease mechanisms in PEX13-related Zellweger spectrum disorders.

Authors:  Paola Borgia; Simona Baldassari; Nicoletta Pedemonte; Ebba Alkhunaizi; Gianluca D'Onofrio; Domenico Tortora; Elisa Calì; Paolo Scudieri; Ganna Balagura; Ilaria Musante; Maria Cristina Diana; Marina Pedemonte; Maria Stella Vari; Michele Iacomino; Antonella Riva; Roberto Chimenz; Giuseppe D Mangano; Mohammad Hasan Mohammadi; Mehran Beiraghi Toosi; Farah Ashrafzadeh; Shima Imannezhad; Ehsan Ghayoor Karimiani; Andrea Accogli; Maria Cristina Schiaffino; Mohamad Maghnie; Miguel Angel Soler; Karl Echiverri; Charles K Abrams; Pasquale Striano; Sara Fortuna; Reza Maroofian; Henry Houlden; Federico Zara; Chiara Fiorillo; Vincenzo Salpietro
Journal:  Orphanet J Rare Dis       Date:  2022-07-19       Impact factor: 4.303

Review 4.  Characterization of Severity in Zellweger Spectrum Disorder by Clinical Findings: A Scoping Review, Meta-Analysis and Medical Chart Review.

Authors:  Mousumi Bose; Christine Yergeau; Yasmin D'Souza; David D Cuthbertson; Melisa J Lopez; Alyssa K Smolen; Nancy E Braverman
Journal:  Cells       Date:  2022-06-10       Impact factor: 7.666

5.  Lactic acidosis and mitochondrial dysfunction in two children with peroxisomal disorders.

Authors:  R D Holmes; K H Moore; J P Ofenstein; P Tsatsos; F L Kiechle
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

6.  A new peroxisomal disorder with enlarged peroxisomes and a specific deficiency of acyl-CoA oxidase (pseudo-neonatal adrenoleukodystrophy).

Authors:  B T Poll-The; F Roels; H Ogier; J Scotto; J Vamecq; R B Schutgens; R J Wanders; C W van Roermund; M J van Wijland; A W Schram
Journal:  Am J Hum Genet       Date:  1988-03       Impact factor: 11.025

Review 7.  Evidence of Oxidative Stress and Secondary Mitochondrial Dysfunction in Metabolic and Non-Metabolic Disorders.

Authors:  Karolina M Stepien; Robert Heaton; Scott Rankin; Alex Murphy; James Bentley; Darren Sexton; Iain P Hargreaves
Journal:  J Clin Med       Date:  2017-07-19       Impact factor: 4.241

8.  Peroxisomes are required for lipid metabolism and muscle function in Drosophila melanogaster.

Authors:  Joseph E Faust; Arvind Manisundaram; Pavlina T Ivanova; Stephen B Milne; James B Summerville; H Alex Brown; Michael Wangler; Michael Stern; James A McNew
Journal:  PLoS One       Date:  2014-06-19       Impact factor: 3.240

  8 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.