Literature DB >> 2427794

Partial deficiency of dihydroxyacetone phosphate acyltransferase activity in both classical and infantile Refsum's diseases.

J T Van Crugten, B Paton, A Poulos.   

Abstract

We measured the activity of dihydroxyacetone phosphate acyltransferase (DHAP-AT) in fibroblasts of controls and patients with classical Refsum's disease (RD), infantile Refsum's disease (IRD) and Zellweger's syndrome (ZS). We confirmed that DHAP-AT activity is severely reduced in ZS fibroblasts and amniocytes. We also demonstrated a partial deficiency of DHAP-AT activity in RD and IRD fibroblast cultures. These diseases are probably distinct but related entities in which peroxisomal biogenesis is affected to varying degrees.

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Year:  1986        PMID: 2427794     DOI: 10.1007/bf01799453

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  20 in total

1.  REFSUM'S SYNDROME: REPORT OF A CASE INCLUDING ELECTRON MICROSCOPIC STUDIES OF THE LIVER.

Authors:  E H KOLODNY; W K HASS; B LANE; W D DRUCKER
Journal:  Arch Neurol       Date:  1965-06

2.  Cerebro-hepato-renal syndrome of Zellweger. A report of eight cases with comments upon the incidence, the liver lesion, and a fault in pipecolic acid metabolism.

Authors:  D M Danks; P Tippett; C Adams; P Campbell
Journal:  J Pediatr       Date:  1975-03       Impact factor: 4.406

3.  Prenatal detection of Zellweger syndrome.

Authors:  R B Schutgens; H S Heymans; R J Wanders; H vd Bosch; G Schrakamp
Journal:  Lancet       Date:  1984-12-08       Impact factor: 79.321

4.  Cerebro-hepato-renal (Zellweger) syndrome and neonatal adrenoleukodystrophy: similarities in phenotype and accumulation of very long chain fatty acids.

Authors:  F R Brown; A J McAdams; J W Cummins; R Konkol; I Singh; A B Moser; H W Moser
Journal:  Johns Hopkins Med J       Date:  1982-12

5.  Deficiency of acyl-CoA: dihydroxyacetone phosphate acyltransferase in patients with Zellweger (cerebro-hepato-renal) syndrome.

Authors:  R B Schutgens; G J Romeyn; R J Wanders; H van den Bosch; G Schrakamp; H S Heymans
Journal:  Biochem Biophys Res Commun       Date:  1984-04-16       Impact factor: 3.575

6.  Deficiency of plasmalogens in the cerebro-hepato-renal (Zellweger) syndrome.

Authors:  H S Heymans; H vd Bosch; R B Schutgens; W H Tegelaers; J U Walther; J Müller-Höcker; P Borst
Journal:  Eur J Pediatr       Date:  1984-04       Impact factor: 3.183

7.  Deficiency of enzymes catalyzing the biosynthesis of glycerol-ether lipids in Zellweger syndrome. A new category of metabolic disease involving the absence of peroxisomes.

Authors:  N S Datta; G N Wilson; A K Hajra
Journal:  N Engl J Med       Date:  1984-10-25       Impact factor: 91.245

8.  Adrenoleukodystrophy: survey of 303 cases: biochemistry, diagnosis, and therapy.

Authors:  H W Moser; A E Moser; I Singh; B P O'Neill
Journal:  Ann Neurol       Date:  1984-12       Impact factor: 10.422

9.  Subcellular localization of acyl coenzyme A: dihydroxyacetone phosphate acyltransferase in rat liver peroxisomes (microbodies).

Authors:  A K Hajra; C L Burke; C L Jones
Journal:  J Biol Chem       Date:  1979-11-10       Impact factor: 5.157

10.  Patterns of Refsum's disease. Phytanic acid oxidase deficiency.

Authors:  A Poulos; A C Pollard; J D Mitchell; G Wise; G Mortimer
Journal:  Arch Dis Child       Date:  1984-03       Impact factor: 3.791

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  4 in total

Review 1.  Clinical approach to inherited peroxisomal disorders.

Authors:  F Poggi-Travert; B Fournier; B T Poll-The; J M Saudubray
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

2.  Prenatal diagnosis of Zellweger syndrome and related disorders: impaired degradation of phytanic acid.

Authors:  A Poulos; C van Crugten; P Sharp; W F Carey; E Robertson; D M Becroft; J M Saudubray; B T Poll-The; E Christensen; N Brandt
Journal:  Eur J Pediatr       Date:  1986-12       Impact factor: 3.183

3.  Autopsy findings in two siblings with infantile Refsum disease.

Authors:  C W Chow; A Poulos; A J Fellenberg; J Christodoulou; D M Danks
Journal:  Acta Neuropathol       Date:  1992       Impact factor: 17.088

4.  Treatment of infantile phytanic acid storage disease: clinical, biochemical and ultrastructural findings in two children treated for 2 years.

Authors:  E F Robertson; A Poulos; P Sharp; J Manson; G Wise; A Jaunzems; R Carter
Journal:  Eur J Pediatr       Date:  1988-02       Impact factor: 3.183

  4 in total

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