Literature DB >> 15186324

The molecular basis of porphyria cutanea tarda in Chile: identification and functional characterization of mutations in the uroporphyrinogen decarboxylase gene.

Pamela Poblete-Gutiérrez1, Manuel Mendez, Tonio Wiederholt, Hans F Merk, Antonio Fontanellas, Carlos Wolff, Jorge Frank.   

Abstract

The porphyrias are heterogeneous disorders arising from predominantly inherited catalytic deficiencies of specific enzymes in heme biosynthesis. Porphyria cutanea tarda (PCT) results from a decreased activity of uroporphyrinogen decarboxylase, the fifth enzyme in heme biosynthesis. The disorder represents the only porphyria that is not exclusively inherited monogenetically. In PCT, at least two different types can be distinguished: acquired/sporadic (type I) PCT, in which the enzymatic deficiency is limited to the liver and inherited/familial (type II) PCT, which is inherited as an autosomal dominant trait with a decrease of enzymatic activity in all tissues. In an effort to characterize the molecular basis of PCT in Chile, we identified eight mutations in 18 previously unclassified PCT families by polymerase chain reaction, heteroduplex analysis, and automated sequencing. To study the role of these mutations in disease causality, in vitro expression of all novel missense mutations was studied. Our results indicate that the frequency of familial PCT in Chile is approximately 50%, thus, to our knowledge, representing the highest incidence of familial PCT reported to date. The data further emphasize the molecular heterogeneity in type II PCT and demonstrate the advantages of molecular genetic techniques as a diagnostic tool and in the detection of clinically asymptomatic mutation carriers.

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Year:  2004        PMID: 15186324     DOI: 10.1111/j.0906-6705.2004.00163.x

Source DB:  PubMed          Journal:  Exp Dermatol        ISSN: 0906-6705            Impact factor:   3.960


  2 in total

Review 1.  [Porphyria cutanea tara].

Authors:  H F Merk
Journal:  Hautarzt       Date:  2016-03       Impact factor: 0.751

2.  Identification and characterization of novel uroporphyrinogen decarboxylase gene mutations in a large series of porphyria cutanea tarda patients and relatives.

Authors:  C Badenas; J To-Figueras; J D Phillips; C A Warby; C Muñoz; C Herrero
Journal:  Clin Genet       Date:  2009-04       Impact factor: 4.438

  2 in total

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