Literature DB >> 3289796

Clinical biochemistry of peroxisomal disorders.

E Kaiser1, R Kramar.   

Abstract

Peroxisomes have been shown to participate in a variety of pathological processes. Peroxisomal anomalities are central features of Zellweger's cerebro-hepato-renal syndrome, neonatal adrenoleukodystrophy, infantile Refsum's disease and several other genetic metabolic disorders (pseudo-Zellweger syndrome, Leber congenital amaurosis, cerebrotendinous xanthomatosis, rhizomelic chondrodysplasia punctata). In disorders with general loss of peroxisomal functions (Zellweger syndrome, neonatal adrenoleukodystrophy, infantile Refsum's disease) an accumulation of very long-chain fatty acids and pathological bile acids are found. Patients have a defective synthesis of plasmalogens and show increased excretion of dicarboxylic acids of medium chain length and of pipecolic acid in the urine. These anomalities which are due to the lack of peroxisomal enzymes, supply the basis for clinical laboratory tests. The study of these peroxisomal disorders has presented valuable information on the normal function of peroxisomes.

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Year:  1988        PMID: 3289796     DOI: 10.1016/0009-8981(88)90357-9

Source DB:  PubMed          Journal:  Clin Chim Acta        ISSN: 0009-8981            Impact factor:   3.786


  2 in total

1.  Pathology of hepatic peroxisomes and mitochondria in patients with peroxisomal disorders.

Authors:  J L Hughes; A Poulos; E Robertson; C W Chow; L J Sheffield; J Christodoulou; R F Carter
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1990

2.  A model-based approach to assess the exposure-response relationship of Lorenzo's oil in adrenoleukodystrophy.

Authors:  Mariam A Ahmed; Reena V Kartha; Richard C Brundage; James Cloyd; Cynthia Basu; Bradley P Carlin; Richard O Jones; Ann B Moser; Ali Fatemi; Gerald V Raymond
Journal:  Br J Clin Pharmacol       Date:  2016-04-03       Impact factor: 4.335

  2 in total

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