Literature DB >> 27704751

Identification of FECH gene multiple variations in two Chinese patients with erythropoietic protoporphyria and a review.

Zhang-Biao Long1, Yong-Wei Wang2,3, Chen Yang1, Gang Liu3, Ya-Li Du1, Guang-Jun Nie3, Yan-Zhong Chang2, Bing Han1.   

Abstract

Erythropoietic protoporphyria (EPP), an autosomal dominant disease, is caused by partial deficiency of ferrochelatase (FECH), which catalyzes the terminal step of heme biosynthesis because of loss-of-function mutations in the FECH gene. To date, only a few cases have been described in Asia. In this study, we describe the clinical features of two Chinese patients with EPP, with diagnosis confirmed by the increase of free protoporphyrin in erythrocytes, detection of plasma fluorescence peak at 630-634 nm, and analysis of FECH gene mutations. Using gene scanning, we identified a small deletion in the FECH gene (c.973 delA) in one proband (patient A) and a pathogenic FECH mutation (c.1232 G>T) in the other (patient B) and also observed some nucleotide variations (c.798 C>G, c.921 A>G, IVS1-23 C>T, IVS3+23 A>G, IVS9+35 C>T, and IVS3-48 T>C) in these patients. The family pedigree of patient A was then established by characterization of the genotype of the patient's relatives. We also analyzed the potential perniciousness of the missense mutation with bioinformatic software, Polyphen and Sift. In summary, Chinese EPP patients have similar manifestations to those of Caucasians, and identification of the Chinese FECH gene mutations expands the FECH genotypic spectrum and may contribute to genetic counseling.

Entities:  

Keywords:  Chinese patients; Clinical manifestation; Erythropoietic protoporphyria; Ferrochelatase; Missense mutations

Mesh:

Substances:

Year:  2016        PMID: 27704751      PMCID: PMC5064175          DOI: 10.1631/jzus.B1600085

Source DB:  PubMed          Journal:  J Zhejiang Univ Sci B        ISSN: 1673-1581            Impact factor:   3.066


  27 in total

1.  An alpha-melanocyte-stimulating hormone analogue in erythropoietic protoporphyria.

Authors:  Juergen Harms; Stephan Lautenschlager; Christoph E Minder; Elisabeth I Minder
Journal:  N Engl J Med       Date:  2009-01-15       Impact factor: 91.245

2.  DNA-based diagnosis of erythropoietic protoporphyria in two families and the frequency of a low-expression FECH allele in a Chinese population.

Authors:  Kin-Chong Lau; Ching-Wan Lam; Bonnie Fong; Tak-Shing Siu; Sidney Tam
Journal:  Clin Chim Acta       Date:  2008-10-10       Impact factor: 3.786

3.  C-terminal deletions in the ALAS2 gene lead to gain of function and cause X-linked dominant protoporphyria without anemia or iron overload.

Authors:  Sharon D Whatley; Sarah Ducamp; Laurent Gouya; Bernard Grandchamp; Carole Beaumont; Michael N Badminton; George H Elder; S Alexander Holme; Alexander V Anstey; Michelle Parker; Anne V Corrigall; Peter N Meissner; Richard J Hift; Joanne T Marsden; Yun Ma; Giorgina Mieli-Vergani; Jean-Charles Deybach; Hervé Puy
Journal:  Am J Hum Genet       Date:  2008-09-04       Impact factor: 11.025

4.  Homozygous deletion of HFE produces a phenotype similar to the HFE p.C282Y/p.C282Y genotype.

Authors:  Gérald Le Gac; Isabelle Gourlaouen; Christophe Ronsin; Vanna Géromel; Anne Bourgarit; Nathalie Parquet; Sylvia Quemener; Cédric Le Maréchal; Jian-Min Chen; Claude Férec
Journal:  Blood       Date:  2008-09-22       Impact factor: 22.113

Review 5.  Erythropoietic protoporphyria.

Authors:  T M Cox
Journal:  J Inherit Metab Dis       Date:  1997-06       Impact factor: 4.982

6.  Long-term observational study of afamelanotide in 115 patients with erythropoietic protoporphyria.

Authors:  G Biolcati; E Marchesini; F Sorge; L Barbieri; X Schneider-Yin; E I Minder
Journal:  Br J Dermatol       Date:  2015-04-30       Impact factor: 9.302

Review 7.  Porphyrias: A 2015 update.

Authors:  Zoubida Karim; Said Lyoumi; Gael Nicolas; Jean-Charles Deybach; Laurent Gouya; Hervé Puy
Journal:  Clin Res Hepatol Gastroenterol       Date:  2015-07-02       Impact factor: 2.947

8.  Afamelanotide for Erythropoietic Protoporphyria.

Authors:  Janneke G Langendonk; Manisha Balwani; Karl E Anderson; Herbert L Bonkovsky; Alexander V Anstey; D Montgomery Bissell; Joseph Bloomer; Chris Edwards; Norbert J Neumann; Charles Parker; John D Phillips; Henry W Lim; Iltefat Hamzavi; Jean-Charles Deybach; Raili Kauppinen; Lesley E Rhodes; Jorge Frank; Gillian M Murphy; Francois P J Karstens; Eric J G Sijbrands; Felix W M de Rooij; Mark Lebwohl; Hetanshi Naik; Colin R Goding; J H Paul Wilson; Robert J Desnick
Journal:  N Engl J Med       Date:  2015-07-02       Impact factor: 91.245

9.  Crystal structure of ferrochelatase: the terminal enzyme in heme biosynthesis.

Authors:  S Al-Karadaghi; M Hansson; S Nikonov; B Jönsson; L Hederstedt
Journal:  Structure       Date:  1997-11-15       Impact factor: 5.006

10.  The difficult clinical diagnosis of erythropoietic protoporphyria.

Authors:  S Wahlin; Y Floderus; A-M Ros; U Broomé; P Harper
Journal:  Physiol Res       Date:  2006       Impact factor: 1.881

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1.  Identification of four hub genes in venous thromboembolism via weighted gene coexpression network analysis.

Authors:  Guoju Fan; Zhihai Jin; Kaiqiang Wang; Huitang Yang; Jun Wang; Yankui Li; Bo Chen; Hongwei Zhang
Journal:  BMC Cardiovasc Disord       Date:  2021-12-03       Impact factor: 2.298

  1 in total

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