Literature DB >> 23153003

Psychological well-being and family satisfaction levels five years after being confirmed as a carrier of the Machado-Joseph disease mutation.

Carlos Gonzalez1, Elisabete Gomes, Nadiya Kazachkova, Conceição Bettencourt, Mafalda Raposo, Teresa Taylor Kay, Patrick MacLeod, João Vasconcelos, Manuela Lima.   

Abstract

The present study on long-term outcome of presymptomatic testing for Machado-Joseph disease (MJD) aimed to evaluate the psychological well-being and the familial satisfaction of subjects that 5 years prior received an unfavorable result in the predictive testing (PT). The study included 47 testees of Azorean origin (23 from the island of Flores and 24 from S. Miguel) that completed the fourth evaluation session of the MJD protocol, and undertook a neurological examination at the moment of participation in the study. Nearly 50% of testees were symptomatic at the time of the study. Psychological well-being of the 47 participants was evaluated using the Psychological General Well-Being Index (PGWB). The family satisfaction scale by adjectives was applied to obtain information on family dynamics. The average PGWB score of the total participants was of 73.3, a value indicative of psychological well-being. Nearly half of the testees presented scores indicating psychological well-being, whereas scores indicating moderate (28.9%) or severe (23.7%) stress were found in the remaining. The average score in the PGWB scale was lower in symptomatic than in asymptomatic subjects; moreover, the distinct distribution of the well-being categories seen in the two groups shows an impact of the appearance of first symptoms on the psychological state. Motives for undertaking the test, provided 5 years prior, failed to show an impact in well-being. The average score for familial satisfaction was of 134, a value compatible with high familial satisfaction, which represented the most frequent category (59.6%). Results demonstrate that well-being and family satisfaction need to be monitored in confirmed carriers of the MJD mutation. The inclusion of acceptance studies, after PT, as well as the development of acceptance training actions, should be of major importance to anticipate the possibility of psychological damage.

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Year:  2012        PMID: 23153003      PMCID: PMC3501113          DOI: 10.1089/gtmb.2011.0370

Source DB:  PubMed          Journal:  Genet Test Mol Biomarkers        ISSN: 1945-0257


  16 in total

1.  Prenatal diagnosis of Machado-Joseph disease by direct mutation analysis.

Authors:  J Sequeiros; P Maciel; F Taborda; S Lêdo; J C Rocha; A Lopes; F Reto; A M Fortuna; M Rousseau; M Fleming; P Coutinho; G A Rouleau; C S Jorge
Journal:  Prenat Diagn       Date:  1998-06       Impact factor: 3.050

2.  Short-term psychological impact of predictive testing for Machado-Joseph disease: depression and anxiety levels in individuals at risk from the Azores (Portugal).

Authors:  Carlos Gonzalez; Manuela Lima; Teresa Kay; Catarina Silva; Cristina Santos; Jorge Santos
Journal:  Community Genet       Date:  2004

3.  CAG expansions in a novel gene for Machado-Joseph disease at chromosome 14q32.1.

Authors:  Y Kawaguchi; T Okamoto; M Taniwaki; M Aizawa; M Inoue; S Katayama; H Kawakami; S Nakamura; M Nishimura; I Akiguchi
Journal:  Nat Genet       Date:  1994-11       Impact factor: 38.330

4.  Preimplantation genetic diagnosis of spinocerebellar ataxia 3 by (CAG)(n) repeat detection.

Authors:  M Drüsedau; J C F M Dreesen; C De Die-Smulders; K Hardy; M Bras; J C M Dumoulin; J L H Evers; H J M Smeets; J P M Geraedts; J Herbergs
Journal:  Mol Hum Reprod       Date:  2004-01       Impact factor: 4.025

5.  Analysis of segregation patterns in Machado-Joseph disease pedigrees.

Authors:  Conceição Bettencourt; Cristina Santos; Teresa Kay; João Vasconcelos; Manuela Lima
Journal:  J Hum Genet       Date:  2008-08-09       Impact factor: 3.172

Review 6.  Dominantly inherited ataxias: lessons learned from Machado-Joseph disease/spinocerebellar ataxia type 3.

Authors:  Henry L Paulson
Journal:  Semin Neurol       Date:  2007-04       Impact factor: 3.420

7.  Correlation between CAG repeat length and clinical features in Machado-Joseph disease.

Authors:  P Maciel; C Gaspar; A L DeStefano; I Silveira; P Coutinho; J Radvany; D M Dawson; L Sudarsky; J Guimarães; J E Loureiro
Journal:  Am J Hum Genet       Date:  1995-07       Impact factor: 11.025

Review 8.  Caring for Machado-Joseph disease: current understanding and how to help patients.

Authors:  Anelyssa D'Abreu; Marcondes C França; Henry L Paulson; Iscia Lopes-Cendes
Journal:  Parkinsonism Relat Disord       Date:  2009-10-06       Impact factor: 4.891

9.  Long-term outcome of presymptomatic testing in Huntington disease.

Authors:  Marcela Gargiulo; Séverine Lejeune; Marie-Laure Tanguy; Khadija Lahlou-Laforêt; Anne Faudet; David Cohen; Josué Feingold; Alexandra Durr
Journal:  Eur J Hum Genet       Date:  2008-08-20       Impact factor: 4.246

10.  Psychological distress in the 5-year period after predictive testing for Huntington's disease.

Authors:  Marleen Decruyenaere; Gerry Evers-Kiebooms; Trees Cloostermans; Andrea Boogaerts; Koen Demyttenaere; René Dom; Jean Pierre Fryns
Journal:  Eur J Hum Genet       Date:  2003-01       Impact factor: 4.246

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  6 in total

1.  Choosing not to know: accounts of non-engagement with pre-symptomatic testing for Machado-Joseph disease.

Authors:  Álvaro Mendes; Milena Paneque; Angus Clarke; Jorge Sequeiros
Journal:  Eur J Hum Genet       Date:  2018-12-20       Impact factor: 4.246

2.  Life paths of patients with transthyretin-related familial amyloid polyneuropathy Val30Met: a descriptive study.

Authors:  Alice Lopes; Alexandra Sousa; Isabel Fonseca; Margarida Branco; Carla Rodrigues; Teresa Coelho; Jorge Sequeiros; Paula Freitas
Journal:  J Community Genet       Date:  2017-10-19

3.  Genetic counseling and presymptomatic testing programs for Machado-Joseph Disease: lessons from Brazil and Portugal.

Authors:  Lavínia Schuler-Faccini; Claudio Maria Osorio; Flavia Romariz; Milena Paneque; Jorge Sequeiros; Laura Bannach Jardim
Journal:  Genet Mol Biol       Date:  2014-03       Impact factor: 1.771

4.  Mid- and long-term anxiety levels associated with presymptomatic testing of Huntington's disease, Machado-Joseph disease, and familial amyloid polyneuropathy.

Authors:  Susana Lêdo; Ângela Leite; Teresa Souto; Maria A Dinis; Jorge Sequeiros
Journal:  Braz J Psychiatry       Date:  2016-02-05       Impact factor: 2.697

Review 5.  Genetic counseling and testing practices for late-onset neurodegenerative disease: a systematic review.

Authors:  Ashley Crook; Chris Jacobs; Toby Newton-John; Rosie O'Shea; Alison McEwen
Journal:  J Neurol       Date:  2021-03-01       Impact factor: 6.682

6.  Short Communication: Restrictions in care following the COVID-19 pandemic severely impacted Machado-Joseph disease patients: a study in the Azores Islands, Portugal.

Authors:  Daniela Couto; Liliana Sousa; Jorge Sequeiros; Manuela Lima; Álvaro Mendes
Journal:  J Community Genet       Date:  2022-07-18
  6 in total

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