Literature DB >> 19084401

Psychosocial impact of presymptomatic genetic testing for transthyretin amyloidotic polyneuropathy.

Anita Graceffa1, Massimo Russo, Gian Luca Vita, Antonio Toscano, Roberto Dattola, Corrado Messina, Giuseppe Vita, Anna Mazzeo.   

Abstract

Presymptomatic genetic testing of an untreatable disease raises clinical, ethical, legal and psychosocial questions. Investigations in specific disorders are needed to help in understanding the motivation for and the impact of genetic testing in the lives of persons at risk for these diseases. Here, we performed a longitudinal study to investigate the psychological consequences of presymptomatic genetic testing on people at risk for transthyretin-related familial amyloidotic polyneuropathy (TTR-FAP). The aim of the present study was to provide possible guidelines for genetic counselling and psychosocial support. Impact of Event Scale Revised (IES-R), Hospital Anxiety and Depression Scale (HADS) and SF-36 questionnaires were administered to 18 asymptomatic subjects before, immediately after communication of the genetic test result and after 3, 6 and 26 months. Our findings showed evidence of anxiety, depression, avoidance of the disease, and psychological distress, especially for women, including those with a negative genetic test result ("survivor guilt"). A psychological support has to be provided before and continued at long term after presymptomatic genetic testing for TTR-FAP in people with positive result as well as in those with negative result.

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Year:  2008        PMID: 19084401     DOI: 10.1016/j.nmd.2008.09.017

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  9 in total

1.  Death anxiety and symbolic immortality in relatives at risk for familial amyloid polyneuropathy type I (FAP I, ATTR V30M).

Authors:  Paula I Santos; Eurico Figueiredo; Inês Gomes; Jorge Sequeiros
Journal:  J Genet Couns       Date:  2010-08-03       Impact factor: 2.537

2.  Phenome-wide association study of TTR and RBP4 genes in 361,194 individuals reveals novel insights in the genetics of hereditary and wildtype transthyretin amyloidoses.

Authors:  Antonella De Lillo; Flavio De Angelis; Marco Di Girolamo; Marco Luigetti; Sabrina Frusconi; Dario Manfellotto; Maria Fuciarelli; Renato Polimanti
Journal:  Hum Genet       Date:  2019-10-29       Impact factor: 4.132

3.  Management of asymptomatic gene carriers of transthyretin familial amyloid polyneuropathy.

Authors:  Hartmut H-J Schmidt; Fabio Barroso; Alejandra González-Duarte; Isabel Conceição; Laura Obici; Denis Keohane; Leslie Amass
Journal:  Muscle Nerve       Date:  2016-09       Impact factor: 3.217

4.  Transthyretin-Related Familial Amyloid Polyneuropathy (TTR-FAP): A Single-Center Experience in Sicily, an Italian Endemic Area.

Authors:  Anna Mazzeo; Massimo Russo; Gianluca Di Bella; Fabio Minutoli; Claudia Stancanelli; Luca Gentile; Sergio Baldari; Scipione Carerj; Antonio Toscano; Giuseppe Vita
Journal:  J Neuromuscul Dis       Date:  2015-07-22

5.  Recommendations for pre-symptomatic genetic testing for hereditary transthyretin amyloidosis in the era of effective therapy: a multicenter Italian consensus.

Authors:  M Grandis; L Obici; M Luigetti; C Briani; F Benedicenti; G Bisogni; M Canepa; F Cappelli; C Danesino; G M Fabrizi; S Fenu; G Ferrandes; C Gemelli; F Manganelli; A Mazzeo; L Melchiorri; F Perfetto; L G Pradotto; P Rimessi; G Tini; S Tozza; L Trevisan; D Pareyson; P Mandich
Journal:  Orphanet J Rare Dis       Date:  2020-12-14       Impact factor: 4.123

6.  Psychosocial burden and professional and social support in patients with hereditary transthyretin amyloidosis (ATTRv) and their relatives in Italy.

Authors:  Lorenza Magliano; Laura Obici; Claudia Sforzini; Anna Mazzeo; Massimo Russo; Francesco Cappelli; Silvia Fenu; Marco Luigetti; Matteo Tagliapietra; Chiara Gemelli; Luca Leonardi; Stefano Tozza; Luca Guglielmo Pradotto; Giulia Citarelli; Alessandro Mauro; Fiore Manganelli; Giovanni Antonini; Marina Grandis; Gian Maria Fabrizi; Mario Sabatelli; Davide Pareyson; Federico Perfetto; Giampaolo Merlini; Giuseppe Vita
Journal:  Orphanet J Rare Dis       Date:  2021-04-07       Impact factor: 4.123

7.  "There are days I wish it wasn't there, and there's days I realize I'm lucky": A qualitative study of psychological sequelae to the implantable cardioverter defibrillator as a treatment for the prevention of sudden cardiac death in arrhythmogenic right ventricular cardiomyopathy.

Authors:  Holly Etchegary; Daryl Pullman; Sean P Connors; Charlene Simmonds; Terry-Lynn Young; Kathy A Hodgkinson
Journal:  JRSM Cardiovasc Dis       Date:  2017-03-08

Review 8.  Recommendations for presymptomatic genetic testing and management of individuals at risk for hereditary transthyretin amyloidosis.

Authors:  Laura Obici; Jan B Kuks; Juan Buades; David Adams; Ole B Suhr; Teresa Coelho; Theodore Kyriakides
Journal:  Curr Opin Neurol       Date:  2016-02       Impact factor: 5.710

9.  Health-related quality of life in hereditary transthyretin amyloidosis polyneuropathy: a prospective, observational study.

Authors:  Mónica Inês; Teresa Coelho; Isabel Conceição; Lara Ferreira; Mamede de Carvalho; João Costa
Journal:  Orphanet J Rare Dis       Date:  2020-03-06       Impact factor: 4.123

  9 in total

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