Literature DB >> 24997713

Seven Novel Mutations in Bulgarian Patients with Acute Hepatic Porphyrias (AHP).

Sonya Dragneva1, Monika Szyszka-Niagolov, Aneta Ivanova, Lyudmila Mateva, Rumiko Izumi, Yoko Aoki, Yoichi Matsubara.   

Abstract

Acute intermittent porphyria (AIP), variegate porphyria (VP), and hereditary coproporphyria (HCP) are caused by mutations in the hydroxymethylbilane synthase (HMBS), protoporphyrinogen oxidase (PPOX), and coproporphyrinogen oxidase (CPOX) genes, respectively. This study aimed to identify mutations in seven Bulgarian families with AIP, six with VP, and one with HCP. A total of 33 subjects, both symptomatic (n = 21) and asymptomatic (n = 12), were included in this study. The identification of mutations was performed by direct sequencing of all the coding exons of the corresponding enzymes in the probands. The available relatives were screened for the possible mutations. A total of six different mutations in HMBS were detected in all seven families with AIP, three of which were previously described: c.76C>T [p.R26C] in exon 3, c.287C>T [p.S96F] in exon 7, and c.445C>T [p.R149X] in exon 9. The following three novel HMBS mutations were found: c.345-2A>C in intron 7-8, c.279-280insAT in exon 7, and c.887delC in exon 15. A total of three different novel mutations were identified in the PPOX gene in the VP families: c.441-442delCA in exon 5, c.917T>C [p.L306P] in exon 9, and c.1252T>C [p.C418R] in exon 12. A novel nonsense mutation, c.364G>T [p.E122X], in exon 1 of the CPOX gene was identified in the HCP family. This study, which identified mutations in Bulgarian families with AHP for the first time, established seven novel mutation sites. Seven latent carriers were also diagnosed and, therefore, were able to receive crucial counseling to prevent attacks.

Entities:  

Year:  2014        PMID: 24997713      PMCID: PMC4221299          DOI: 10.1007/8904_2014_320

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  27 in total

Review 1.  ACP Best Practice No 165: front line tests for the investigation of suspected porphyria.

Authors:  A C Deacon; G H Elder
Journal:  J Clin Pathol       Date:  2001-07       Impact factor: 3.411

Review 2.  Modern diagnosis and management of the porphyrias.

Authors:  Shigeru Sassa
Journal:  Br J Haematol       Date:  2006-09-04       Impact factor: 6.998

3.  Marked geographic aggregation of acute intermittent porphyria families carrying mutation Q180X in Venezuelan populations, with description of further mutations.

Authors:  Irene Paradisi; Sergio Arias
Journal:  J Inherit Metab Dis       Date:  2010-10-27       Impact factor: 4.982

4.  Molecular epidemiology and diagnosis of PBG deaminase gene defects in acute intermittent porphyria.

Authors:  H Puy; J C Deybach; J Lamoril; A M Robreau; V Da Silva; L Gouya; B Grandchamp; Y Nordmann
Journal:  Am J Hum Genet       Date:  1997-06       Impact factor: 11.025

5.  Acute intermittent porphyria in Finland: 19 mutations in the porphobilinogen deaminase gene.

Authors:  R Kauppinen; S Mustajoki; H Pihlaja; L Peltonen; P Mustajoki
Journal:  Hum Mol Genet       Date:  1995-02       Impact factor: 6.150

6.  Plasma fluorescence scanning and fecal porphyrin analysis for the diagnosis of variegate porphyria: precise determination of sensitivity and specificity with detection of protoporphyrinogen oxidase mutations as a reference standard.

Authors:  Richard J Hift; Brandon P Davidson; Cornelis van der Hooft; Doreen M Meissner; Peter N Meissner
Journal:  Clin Chem       Date:  2004-02-19       Impact factor: 8.327

7.  Structure of human porphobilinogen deaminase at 2.8 A: the molecular basis of acute intermittent porphyria.

Authors:  Raj Gill; Simon E Kolstoe; Fiyaz Mohammed; Abeer Al D-Bass; Julie E Mosely; Mohammed Sarwar; Jonathan B Cooper; Stephen P Wood; Peter M Shoolingin-Jordan
Journal:  Biochem J       Date:  2009-04-28       Impact factor: 3.857

Review 8.  Porphyria in Sweden.

Authors:  S Thunell; Y Floderus; A Henrichson; P Harper
Journal:  Physiol Res       Date:  2006       Impact factor: 1.881

Review 9.  May 2006 update in porphobilinogen deaminase gene polymorphisms and mutations causing acute intermittent porphyria: comparison with the situation in Slavic population.

Authors:  M Hrdinka; H Puy; P Martasek
Journal:  Physiol Res       Date:  2006       Impact factor: 1.881

10.  Partial protoporphyrinogen oxidase (PPOX) gene deletions, due to different Alu-mediated mechanisms, identified by MLPA analysis in patients with variegate porphyria.

Authors:  Michela Barbaro; Maire Kotajärvi; Pauline Harper; Ylva Floderus
Journal:  Orphanet J Rare Dis       Date:  2013-01-16       Impact factor: 4.123

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