Literature DB >> 12699245

Molecular characterization of porphyrias in Italy: a diagnostic flow-chart.

F Martinez di Montemuros1, E Di Pierro, E Patti, D Tavazzi, M G Danielli, G Biolcati, E Rocchi, M D Cappellini.   

Abstract

The porphyrias are disorders associated with inherited or acquired enzyme deficiencies in the heme biosynthetic pathway. The differential diagnosis is often difficult since the phenotype is very similar in some forms and the biochemical tests are not commonly available. Here we provide an update on the molecular diagnosis of porphyrias in Italy and a flow-chart to facilitate the identification of mutations in heme biosynthetic genes. The molecular analysis has allowed us to identify the molecular defect underlying the disease in 66 probands with different porphyrias [acute intermittent porphyria (AIP), variegate porphyria (VP), porphyria cutanea tarda (PCT), erythropoietic protoporphyria (EPP)]. No Italian patients with defects in coproporphyrinogen oxidise (CPOX) gene, responsible for hereditary coproporphyria (HCP), have been detected. The molecular characterization has been extended to 115 relatives with the identification of 55 asymptomatic mutation carriers and 60 normal subjects. We have so far identified 50 different mutations among 4 genes associated with the most common porphyrias showing a high molecular heterogeneity: 22 in the hydroxymethylbilane synthase (HMBS) gene (AIP), 7 in the protoporphyrinogen oxidase (PPOX) gene (VP), 16 in the uroporphyrinogen decarboxylase (UROD) gene (PCT) and 5 in the ferrochelatase (FECH) gene (EPP). Among the 50 molecular defects, 29 seem to be restricted to the Italian population.

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Year:  2002        PMID: 12699245

Source DB:  PubMed          Journal:  Cell Mol Biol (Noisy-le-grand)        ISSN: 0145-5680            Impact factor:   1.770


  4 in total

1.  The porphyrias: pathophysiology.

Authors:  Antonello Pietrangelo
Journal:  Intern Emerg Med       Date:  2010-10       Impact factor: 3.397

2.  Paralytic ileus and liver failure--an unusual presentation of advanced erythropoietic protoporphyria.

Authors:  Simone Negrini; Gabriele Zoppoli; Maurizio Setti; Maria Domenica Cappellini; Francesco Indiveri
Journal:  Dig Dis Sci       Date:  2008-11-14       Impact factor: 3.199

3.  Clinic and genetic evaluation of variegate porphyria (VP) in a large family from the Balearic Islands.

Authors:  A Bonnin; A Picornell; J Orfila; J A Castro; M M Ramon
Journal:  J Inherit Metab Dis       Date:  2009-02-20       Impact factor: 4.982

4.  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

  4 in total

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