Literature DB >> 32043565

Genetics of Wilson disease and Wilson-like phenotype in a clinical series from eastern Spain.

Ana Sánchez-Monteagudo1,2, María Álvarez-Sauco3, Isabel Sastre4, Irene Martínez-Torres4, Vincenzo Lupo1,2, Marina Berenguer2,5,6, Carmen Espinós1,2.   

Abstract

Wilson's disease (WD) is an autosomal recessive disorder caused by ATP7B mutations. Subjects with only one mutation may show clinical signs and individuals with biallelic changes may remain asymptomatic. We aimed to achieve a conclusive genetic diagnosis for 34 patients clinically diagnosed of WD. Genetic analysis comprised from analysis of exons to WES (whole exome sequencing), including promoter, introns, UTRs (untranslated regions), besides of study of large deletions/duplications by MLPA (multiplex ligation-dependent probe amplification). Biallelic ATP7B mutations were identified in 30 patients, so that four patients were analyzed using WES. Two affected siblings resulted to be compound heterozygous for mutations in CCDC115, which is involved in a form of congenital disorder of glycosylation. In sum, the majority of patients with a WD phenotype carry ATP7B mutations. However, if genetic diagnosis is not achieved, additional genes should be considered because other disorders may mimic WD.
© 2020 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  zzm321990ATP7B gene; zzm321990CCDC115 gene; Wilson's disease; Wilson-like phenotype; genetic diagnosis; targeted next-generation sequencing; whole exome sequencing

Mesh:

Substances:

Year:  2020        PMID: 32043565     DOI: 10.1111/cge.13719

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

1.  Wilson disease: revision of diagnostic criteria in a clinical series with great genetic homogeneity.

Authors:  Luis García-Villarreal; Andrea Hernández-Ortega; Ana Sánchez-Monteagudo; Luis Peña-Quintana; Teresa Ramírez-Lorenzo; Marta Riaño; Raquel Moreno-Pérez; Alberto Monescillo; Daniel González-Santana; Ildefonso Quiñones; Almudena Sánchez-Villegas; Vicente Olmo-Quintana; Paloma Garay-Sánchez; Carmen Espinós; Jesús M González; Antonio Tugores
Journal:  J Gastroenterol       Date:  2020-11-07       Impact factor: 7.527

Review 2.  Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?

Authors:  Carmen Espinós; Peter Ferenci
Journal:  JHEP Rep       Date:  2020-04-18

3.  NR4A2 Mutations Can Cause Intellectual Disability and Language Impairment With Persistent Dystonia-Parkinsonism.

Authors:  Silvia Jesús; Isabel Hinarejos; Fátima Carrillo; Dolores Martínez-Rubio; Daniel Macías-García; Ana Sánchez-Monteagudo; Astrid Adarmes; Vincenzo Lupo; Belén Pérez-Dueñas; Pablo Mir; Carmen Espinós
Journal:  Neurol Genet       Date:  2021-01-21

4.  Mutations, Genes, and Phenotypes Related to Movement Disorders and Ataxias.

Authors:  Dolores Martínez-Rubio; Isabel Hinarejos; Paula Sancho; Nerea Gorría-Redondo; Raquel Bernadó-Fonz; Cristina Tello; Clara Marco-Marín; Itxaso Martí-Carrera; María Jesús Martínez-González; Ainhoa García-Ribes; Raquel Baviera-Muñoz; Isabel Sastre-Bataller; Irene Martínez-Torres; Anna Duat-Rodríguez; Patrícia Janeiro; Esther Moreno; Leticia Pías-Peleteiro; Mar O'Callaghan Gordo; Ángeles Ruiz-Gómez; Esteban Muñoz; Maria Josep Martí; Ana Sánchez-Monteagudo; Candela Fuster; Amparo Andrés-Bordería; Roser Maria Pons; Silvia Jesús-Maestre; Pablo Mir; Vincenzo Lupo; Belén Pérez-Dueñas; Alejandra Darling; Sergio Aguilera-Albesa; Carmen Espinós
Journal:  Int J Mol Sci       Date:  2022-10-06       Impact factor: 6.208

5.  ATP7B variant c.1934T > G p.Met645Arg causes Wilson disease by promoting exon 6 skipping.

Authors:  Daniele Merico; Carl Spickett; Matthew O'Hara; Boyko Kakaradov; Amit G Deshwar; Phil Fradkin; Shreshth Gandhi; Jiexin Gao; Solomon Grant; Ken Kron; Frank W Schmitges; Zvi Shalev; Mark Sun; Marta Verby; Matthew Cahill; James J Dowling; Johan Fransson; Erno Wienholds; Brendan J Frey
Journal:  NPJ Genom Med       Date:  2020-04-08       Impact factor: 8.617

6.  Synonymous mutation in adenosine triphosphatase copper-transporting beta causes enhanced exon skipping in Wilson disease.

Authors:  Marlene Panzer; André Viveiros; Benedikt Schaefer; Nadja Baumgartner; Klaus Seppi; Atbin Djamshidian; Theodor Todorov; William J H Griffiths; Eckart Schott; Markus Schuelke; Dennis Eurich; Albert Friedrich Stättermayer; Adrian Bomford; Pierre Foskett; Julia Vodopiutz; Rudolf Stauber; Elke Pertler; Bernhard Morell; Herbert Tilg; Thomas Müller; Stefan Kiechl; Raul Jimenez-Heredia; Karl Heinz Weiss; Si Houn Hahn; Andreas Janecke; Peter Ferenci; Heinz Zoller
Journal:  Hepatol Commun       Date:  2022-03-10
  6 in total

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