Literature DB >> 30987916

Pathogenesis and clinical features of the acute hepatic porphyrias (AHPs).

Herbert L Bonkovsky1, Natalia Dixon2, Sean Rudnick3.   

Abstract

The acute hepatic porphyrias include four disorders: acute intermittent porphyria [AIP], hereditary coproporphyria [HCP], variegate porphyria [VP], and the rare porphyria due to severe deficiency of ALA dehydratase [ADP]. In the USA, AIP is the most severe and most often symptomatic. AIP, HCP, and VP are due to autosomal dominant genetic abnormalities, in which missense, nonsense, or other mutations of genes of normal hepatic heme biosynthesis, in concert with other environmental, nutritional, hormonal and genetic factors, may lead to a critical deficiency of heme, the end-product of the pathway, in a small but critical 'regulatory pool' within hepatocytes. This deficiency leads to de-repression of the first and normally rate-controlling enzyme of the heme synthetic pathway, delta- or 5-aminolevulinic acid [ALA] synthase-1, and thus to marked up-regulation of this key enzyme and to marked hepatic overproduction of ALA. In addition, except for ADP, there is marked overproduction as well of porphobilinogen [PBG], the intermediate immediately downstream of ALA in the synthetic chain, and, especially in HCP and VP, also porphyrinogens and porphyrins farther down the pathway. The major clinical features of the acute porphyrias are attacks of severe neuropathic-type pain. Pain is felt first and foremost in the abdomen but may also occur in the back, chest, and extremities. Attacks are more common in women than in men [ratio of about 4:1], often accompanied by nausea, vomiting, constipation, tachycardia, and arterial hypertension. Hyponatremia may also occur. Some patients also describe chronic symptoms of pain, anxiety, insomnia, and others.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  5-aminolevulinic acid; 5-aminolevulinic acid synthase; Acute hepatic porphyrias; Acute porphyric attacks; Heme; Hydroxymethylbilane synthase; Porphobilinogen [deaminase]; Porphyrias; Porphyrins

Mesh:

Substances:

Year:  2019        PMID: 30987916      PMCID: PMC6754303          DOI: 10.1016/j.ymgme.2019.03.002

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  28 in total

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Authors:  M Hahn; O S Gildemeister; G L Krauss; J A Pepe; R W Lambrecht; S Donohue; H L Bonkovsky
Journal:  Neurology       Date:  1997-07       Impact factor: 9.910

2.  Acute intermittent porphyria: a study of 50 cases.

Authors:  A GOLDBERG
Journal:  Q J Med       Date:  1959-04

3.  Homozygous acute intermittent porphyria: compound heterozygosity for adjacent base transitions in the same codon of the porphobilinogen deaminase gene.

Authors:  D H Llewellyn; S J Smyth; G H Elder; A C Hutchesson; J M Rattenbury; M F Smith
Journal:  Hum Genet       Date:  1992-04       Impact factor: 4.132

4.  Acute intermittent porphyria: prevalence of mutations in the porphobilinogen deaminase gene in blood donors in France.

Authors:  Y Nordmann; H Puy; V Da Silva; S Simonin; A M Robreau; C Bonaiti; L N Phung; J C Deybach
Journal:  J Intern Med       Date:  1997-09       Impact factor: 8.989

Review 5.  Acute intermittent porphyria. A clinical and biochemical study of 46 patients.

Authors:  J A Stein; D P Tschudy
Journal:  Medicine (Baltimore)       Date:  1970-01       Impact factor: 1.889

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7.  Intravenous heme-albumin in acute intermittent porphyria: evidence for repletion of hepatic hemoproteins and regulatory heme pools.

Authors:  H L Bonkovsky; J F Healey; A N Lourie; G G Gerron
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7.  Severe neuropathic attack in a woman with acute intermittent porphyria: a case report.

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Review 10.  Therapeutic potential of pyrrole and pyrrolidine analogs: an update.

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