Literature DB >> 33914262

Compound heterozygosity for two novel mutations of the SEC23B gene in congenital dyserythropoietic anemia type II.

Shanshan Chen1, Ziwen Guo1, Yongbin Ye1, Shanhong Yang2, Guinian Huang3.   

Abstract

Congenital dyserythropoietic anemia type II (CDA II), a rare genetic disorder, results from SEC23B gene mutations according to previous studies. Here, we present a case of CDA II involving two novel pathogenic mutations of SEC23B that have not previously been reported. The patient suffered from jaundice, tea-colored urine, and weakness. Laboratory data indicated moderately decreased hemoglobin, iron overload, and abnormal erythroblast morphology. Therefore, a diagnosis of CDA II was considered. Peripheral blood samples were used to perform whole exome sequencing, and the results showed compound heterozygosity of the SEC23B gene with the following mutations: c.1162T>A (p.F388I) and c.1603delC (p.R535del). The mutant proteins were predicted to be deleterious and resulted in decreased structural stability. PyMOL software was used to analyze the structural change caused by the p.F388I missense mutation, and the results indicated a deficiency in π-π interactions. In conclusion, our report extends the mutation spectrum of SEC23B in the diagnosis of CDA II.

Entities:  

Keywords:  CDA II; Exome sequencing; Mutation; SEC23B

Year:  2021        PMID: 33914262     DOI: 10.1007/s12185-021-03155-1

Source DB:  PubMed          Journal:  Int J Hematol        ISSN: 0925-5710            Impact factor:   2.490


  16 in total

1.  Localization of the congenital dyserythropoietic anemia II locus to chromosome 20q11.2 by genomewide search.

Authors:  P Gasparini; E Miraglia del Giudice; J Delaunay; A Totaro; M Granatiero; S Melchionda; L Zelante; A Iolascon
Journal:  Am J Hum Genet       Date:  1997-11       Impact factor: 11.025

Review 2.  Advances in the understanding of the congenital dyserythropoietic anaemias.

Authors:  Sunitha N Wickramasinghe; William G Wood
Journal:  Br J Haematol       Date:  2005-11       Impact factor: 6.998

3.  Mutations affecting the secretory COPII coat component SEC23B cause congenital dyserythropoietic anemia type II.

Authors:  Klaus Schwarz; Achille Iolascon; Fatima Verissimo; Nikolaus S Trede; Wyatt Horsley; Wen Chen; Barry H Paw; Karl-Peter Hopfner; Karlheinz Holzmann; Roberta Russo; Maria Rosaria Esposito; Daniela Spano; Luigia De Falco; Katja Heinrich; Brigitte Joggerst; Markus T Rojewski; Silverio Perrotta; Jonas Denecke; Ulrich Pannicke; Jean Delaunay; Rainer Pepperkok; Hermann Heimpel
Journal:  Nat Genet       Date:  2009-06-28       Impact factor: 38.330

4.  Analysis of a cohort of 101 CDAII patients: description of 24 new molecular variants and genotype-phenotype correlations.

Authors:  Paola Bianchi; Klaus Schwarz; Josef Högel; Elisa Fermo; Cristina Vercellati; Regine Grosse; Richard van Wijk; Rob van Zwieten; Wilma Barcellini; Alberto Zanella; Hermann Heimpel
Journal:  Br J Haematol       Date:  2016-07-29       Impact factor: 6.998

5.  Congenital dyserythropoietic anemia.

Authors:  Takahiro Kamiya; Atsushi Manabe
Journal:  Int J Hematol       Date:  2010-09-07       Impact factor: 2.490

6.  Frequency of congenital dyserythropoietic anemias in Europe.

Authors:  Hermann Heimpel; Andreas Matuschek; Momin Ahmed; Brigitte Bader-Meunier; Adriana Colita; Jean Delaunay; Loic Garcon; Florinda Gilsanz; Jeroen Goede; Josef Högel; Elisabeth Kohne; Rosi Leichtle; Juan Munoz; Silverio Perrotta; Carlo Piscopo; Raffaele Renella; Klaus Schwarz; Gabriela Smolenska-Sym; Sunitha Wickramasinghe; Alberto Zanella; Achille Iolascon
Journal:  Eur J Haematol       Date:  2010-07       Impact factor: 2.997

7.  Congenital dyserythropoietic anemia type II (CDAII) is caused by mutations in the SEC23B gene.

Authors:  Paola Bianchi; Elisa Fermo; Cristina Vercellati; Carla Boschetti; Wilma Barcellini; Alessandra Iurlo; Anna Paola Marcello; Pier Giorgio Righetti; Alberto Zanella
Journal:  Hum Mutat       Date:  2009-09       Impact factor: 4.878

8.  Retrospective cohort study of 205 cases with congenital dyserythropoietic anemia type II: definition of clinical and molecular spectrum and identification of new diagnostic scores.

Authors:  Roberta Russo; Antonella Gambale; Concetta Langella; Immacolata Andolfo; Sule Unal; Achille Iolascon
Journal:  Am J Hematol       Date:  2014-07-22       Impact factor: 10.047

9.  Mutational spectrum in congenital dyserythropoietic anemia type II: identification of 19 novel variants in SEC23B gene.

Authors:  Roberta Russo; Maria Rosaria Esposito; Roberta Asci; Antonella Gambale; Silverio Perrotta; Ugo Ramenghi; Gian Luca Forni; Vedat Uygun; Jean Delaunay; Achille Iolascon
Journal:  Am J Hematol       Date:  2010-12       Impact factor: 10.047

Review 10.  Congenital dyserythropoietic anemias: molecular insights and diagnostic approach.

Authors:  Achille Iolascon; Hermann Heimpel; Anders Wahlin; Hannah Tamary
Journal:  Blood       Date:  2013-08-12       Impact factor: 22.113

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