Literature DB >> 513604

New type of hepatic porphyria with porphobilinogen synthase defect and intermittent acute clinical manifestation.

M Doss, R von Tiepermann, J Schneider, H Schmid.   

Abstract

In two young patients with acute hepatic porphyria syndrome and persisting paralyses, which increased in intensity during intermittent occurring crisis, the activity of erythrocyte porphobilinogen synthase (delta-aminolevulinic acid dehydratase) was found to be considerably diminished, below 1% of the value of normal control persons. In contrast, the activity of uroporphyrinogen synthase was normal. Both patients have been excreting high quantities of delta-aminolevulinic acid and porphyrins in urine for years. Lead intoxication has definitively been excluded. Since the relatives also show lower activities in porphobilinogen synthase, the disease of these two patients is probably a new enzymatic type of inherited acute hepatic porphyria, the excretion profile of which is qualitatively completely different from those of the known acute porphyrias. The discovery of this porphyria confirms the theory of overlapping transition in the biochemical and clinical symptoms and analogies among acute hepatic porphyrias.

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Year:  1979        PMID: 513604     DOI: 10.1007/bf01481493

Source DB:  PubMed          Journal:  Klin Wochenschr        ISSN: 0023-2173


  13 in total

1.  Acute lead poisoning: Five cases resulting from self-injection of lead and opium.

Authors:  A D Beattie; J D Briggs; J S Canavan; D Doyle; P J Mullin; A A Watson
Journal:  Q J Med       Date:  1975-04

2.  delta-Aminolevulinic acid: influences on synaptic GABA receptor binding may explain CNS symptoms of porphyria.

Authors:  W E Müller; S H Snyder
Journal:  Ann Neurol       Date:  1977-10       Impact factor: 10.422

Review 3.  Exposure of humans to lead.

Authors:  P B Hammond
Journal:  Annu Rev Pharmacol Toxicol       Date:  1977       Impact factor: 13.820

4.  Studies in lead poisoning. II. Correlation between the ratio of activated to inactivated delta-aminolevulinic acid dehydratase of whole blood and the blood lead level.

Authors:  J L Granick; S Sassa; S Granick; R D Levere; A Kappas
Journal:  Biochem Med       Date:  1973-08

5.  Urinary porphyrin patterns in hepatic porphyrias.

Authors:  M Doss
Journal:  Klin Wochenschr       Date:  1971-08-15

6.  Tyrosinemia with aminolevulinic dehydratase deficiency.

Authors:  J M Lamon; B C Frykholm; D P Tschudy
Journal:  J Pediatr       Date:  1978-02       Impact factor: 4.406

7.  Relationships between acute hepatic porphyrias due to genetic variability of primary enzyme defects and limiting function of uroporphyrinogen synthase.

Authors:  M Doss
Journal:  Int J Biochem       Date:  1978

8.  Hematologic and biochemical studies in a case of lead poisoning.

Authors:  P D Berk; D P Tschudy; L A Shepley; J G Waggoner; N I Berlin
Journal:  Am J Med       Date:  1970-01       Impact factor: 4.965

9.  Porphyrin precursors in acute porphyria.

Authors:  L Eales
Journal:  S Afr Med J       Date:  1978-01-21

10.  Tyrosinemia with acute intermittent porphyria: aminolevulinic acid dehydratase deficiency related to elevated urinary aminolevulinic acid levels.

Authors:  C F Strife; E L Zuroweste; E A Emmett; V N Finelli; H G Petering; H K Berry
Journal:  J Pediatr       Date:  1977-03       Impact factor: 4.406

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  31 in total

1.  Survival of two patients with severe delta-aminolaevulinic acid dehydratase deficiency porphyria.

Authors:  U Gross; S Sassa; T Arndt; M O Doss
Journal:  J Inherit Metab Dis       Date:  2001-02       Impact factor: 4.982

2.  X-ray structure of a putative reaction intermediate of 5-aminolaevulinic acid dehydratase.

Authors:  Peter T Erskine; Leighton Coates; Danica Butler; James H Youell; Amanda A Brindley; Steve P Wood; Martin J Warren; Peter M Shoolingin-Jordan; Jonathan B Cooper
Journal:  Biochem J       Date:  2003-08-01       Impact factor: 3.857

3.  Direct assay of delta-aminolevulinic acid dehydratase in heme biosynthesis for the detection of porphyrias by tandem mass spectrometry.

Authors:  John R Choiniere; C Ronald Scott; Michael H Gelb; Frantisek Turecek
Journal:  Anal Chem       Date:  2010-08-01       Impact factor: 6.986

Review 4.  Hepatic porphyrias in children.

Authors:  G H Elder
Journal:  J Inherit Metab Dis       Date:  1997-06       Impact factor: 4.982

5.  Lead poisoning in inherited delta-aminolevulinic acid dehydratase deficiency.

Authors:  M Doss; F Laubenthal; M Stoeppler
Journal:  Int Arch Occup Environ Health       Date:  1984       Impact factor: 3.015

6.  Hematin therapy in porphyric attacks.

Authors:  C A Pierach; I Bossenmaier; R Cardinal; M Weimer; C J Watson
Journal:  Klin Wochenschr       Date:  1980-08-15

7.  Hereditary tyrosinemia and the heme biosynthetic pathway. Profound inhibition of delta-aminolevulinic acid dehydratase activity by succinylacetone.

Authors:  S Sassa; A Kappas
Journal:  J Clin Invest       Date:  1983-03       Impact factor: 14.808

8.  Lead poisoning as a toxogenetic disease.

Authors:  M Doss; H Baumann; M Lorrek; F Laubenthal; J Schneider
Journal:  Klin Wochenschr       Date:  1984-05-02

9.  The "glucose effect" in acute hepatic porphyrias and in experimental porphyria.

Authors:  M Doss; F Verspohl
Journal:  Klin Wochenschr       Date:  1981-07-01

10.  Porphobilinogen-synthase (delta-aminolevulinic acid dehydratase) deficiency in bone marrow cells of two patients with porphobilinogen-synthase defect acute porphyria.

Authors:  M Doss; R V Tiepermann; J Schneider
Journal:  Klin Wochenschr       Date:  1983-07-15
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