Literature DB >> 30328212

Analysis of the TTR gene in the investigation of amyloidosis: A 25-year single UK center experience.

Dorota Rowczenio1, Candida C Quarta1, Marianna Fontana1, Carol J Whelan1, Ana Martinez-Naharro1, Hadija Trojer1, Anna Baginska1, Stuart M Ferguson1, Janet Gilbertson1, Tamer Rezk1, Sajitha Sachchithanantham1, Shameem Mahmood1, Richa Manwani1, Faye Sharpley1, Ashutosh D Wechalekar1, Philip N Hawkins1, Julian D Gillmore1, Helen J Lachmann1.   

Abstract

Transthyretin amyloidosis (ATTR) is caused by deposition of either wild-type (ATTRwt) or variant (ATTRm) transthyretin. ATTRwt presents with restrictive cardiomyopathy, while ATTRm displays a range of organ involvement. This retrospective analysis includes all patients referred to a single UK center in the last 25 years for clinical and laboratory assessment of known or suspected amyloidosis who underwent TTR gene sequencing. A total of 4459 patients were included in this study; 37% had final diagnosis of ATTR amyloidosis; 27% light chain amyloidosis; 0.7% other types of amyloidosis; 21.3% had no amyloid and 14% had no data. TTR variants were found in 770 (17%) cases; the most prevalent were p.V142I, p.T80A, and p.V50M identified in 42, 25, and 16%, respectively. The median age at referral in each group was: 76 (range 47-93), 66 (40-81), and 45 years (21-86), respectively. Overall 42 rare or novel variants were identified. Forty-two percent patients with ATTRm died at a median age of 73 years (33-89) with a median survival from diagnosis of 50 months. ATTRwt was the final diagnosis in 20% of patients undergoing genetic testing. Our findings of TTR variants in 17% of screened patients highlight the need for routine genetic testing in the evaluation of suspected ATTR amyloidosis.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  TTR gene variants; cardiomyopathy; neuropathy; transthyretin amyloidosis (ATTR); variant (ATTRm) transthyretin amyloidosis; wild-type (ATTRwt) transthyretin amyloidosis

Mesh:

Substances:

Year:  2018        PMID: 30328212     DOI: 10.1002/humu.23669

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  7 in total

1.  Racial and Ethnic Disparities in Transthyretin Cardiac Amyloidosis.

Authors:  Gabriela Spencer-Bonilla; Joyce N Njoroge; Keon Pearson; Ronald M Witteles; Mandar A Aras; Kevin M Alexander
Journal:  Curr Cardiovasc Risk Rep       Date:  2021-04-04

2.  Prevalence and Outcomes of p.Val142Ile TTR Amyloidosis Cardiomyopathy: A Systematic Review.

Authors:  Pranav Chandrashekar; Laith Alhuneafat; Meghan Mannello; Lana Al-Rashdan; Morris M Kim; Jason Dungu; Kevin Alexander; Ahmad Masri
Journal:  Circ Genom Precis Med       Date:  2021-08-31

3.  Molecular Diagnosis of Inherited Cardiac Diseases in the Era of Next-Generation Sequencing: A Single Center's Experience Over 5 Years.

Authors:  Alexandre Janin; Louis Januel; Cécile Cazeneuve; Antoine Delinière; Philippe Chevalier; Gilles Millat
Journal:  Mol Diagn Ther       Date:  2021-05-05       Impact factor: 4.074

4.  Epidemiology of variant transthyretin amyloidosis at a reference center in Argentina.

Authors:  Maria S Saez; Maria A Aguirre; Diego Pérez de Arenaza; Patricia Sorroche; Elsa Nucifora; Maria L Posadas Martinez
Journal:  Mol Genet Genomic Med       Date:  2021-10-20       Impact factor: 2.183

5.  Clinical and Genetic Evaluation of People with or at Risk of Hereditary ATTR Amyloidosis: An Expert Opinion and Consensus on Best Practice in Ireland and the UK.

Authors:  Julian D Gillmore; Mary M Reilly; Caroline J Coats; Rob Cooper; Helen Cox; Mark R E Coyne; Andrew J Green; Ruth McGowan; William E Moody; Philip N Hawkins
Journal:  Adv Ther       Date:  2022-04-13       Impact factor: 4.070

Review 6.  Transthyretin cardiac amyloidosis: A treatable form of heart failure with a preserved ejection fraction.

Authors:  Jan M Griffin; Mathew S Maurer
Journal:  Trends Cardiovasc Med       Date:  2019-12-17       Impact factor: 6.677

7.  Plasmin activity promotes amyloid deposition in a transgenic model of human transthyretin amyloidosis.

Authors:  Ivana Slamova; Rozita Adib; Stephan Ellmerich; Michal R Golos; Janet A Gilbertson; Nicola Botcher; Diana Canetti; Graham W Taylor; Nigel Rendell; Glenys A Tennent; Guglielmo Verona; Riccardo Porcari; P Patrizia Mangione; Julian D Gillmore; Mark B Pepys; Vittorio Bellotti; Philip N Hawkins; Raya Al-Shawi; J Paul Simons
Journal:  Nat Commun       Date:  2021-12-07       Impact factor: 14.919

  7 in total

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