Literature DB >> 28799081

The Psychosocial Impact of Carrying a Debated Variant in the GLA Gene.

Sarah Macklin1,2, Dawn Laney3, Emily Lisi3,4, Andrea Atherton5, Elizabeth Smith3.   

Abstract

The clinical significance of the c.427G>A (p.A143T) variant in GLA is a topic of debate within the lysosomal storage disease community. A review of the literature and published case reports found the clinical impact of the variant to range from classic Fabry symptoms to healthy unaffected males with normal alpha- galactosidase enzyme levels, leaving clinicians unsure of how to manage these individuals. As the number of states testing for Fabry disease on their newborn screening panel has increased, more people with this variant are being identified. The goal of this project was to learn how the uncertainty surrounding the clinical significance of the p.A143T variant affects those with this change. A self-response questionnaire was developed to explore this topic. In addition to evaluating participant feelings, the questionnaire explored individuals' beliefs regarding the pathogenicity of the variant. Results suggest that people have diverse feelings regarding reclassification of the p.A143T variant. Around half of those surveyed reported feeling frustrated by the lack of clear information. Despite the ambiguity regarding the health consequences of this variant, many participants felt that knowing this result helps guide medical management.

Entities:  

Keywords:  Fabry disease; Psychosocial; Variant of uncertain significance

Mesh:

Substances:

Year:  2017        PMID: 28799081     DOI: 10.1007/s10897-017-0139-y

Source DB:  PubMed          Journal:  J Genet Couns        ISSN: 1059-7700            Impact factor:   2.537


  25 in total

1.  Questioning the Pathogenic Role of the GLA p.Ala143Thr "Mutation" in Fabry Disease: Implications for Screening Studies and ERT.

Authors:  W Terryn; R Vanholder; D Hemelsoet; B P Leroy; W Van Biesen; G De Schoenmakere; B Wuyts; K Claes; J De Backer; G De Paepe; A Fogo; M Praet; B Poppe
Journal:  JIMD Rep       Date:  2012-07-29

Review 2.  Animal models of anxiety and depression: how are females different?

Authors:  P Palanza
Journal:  Neurosci Biobehav Rev       Date:  2001-05       Impact factor: 8.989

3.  Social-adaptive and psychological functioning of patients affected by Fabry disease.

Authors:  Dawn Alyssia Laney; Daniel J Gruskin; Paul M Fernhoff; Joseph F Cubells; Opal Y Ousley; Heather Hipp; Ami J Mehta
Journal:  J Inherit Metab Dis       Date:  2010-01-20       Impact factor: 4.982

4.  Preimplantation genetic diagnosis: patients' experiences and attitudes.

Authors:  S A Lavery; R Aurell; C Turner; C Castello; A Veiga; P N Barri; R M Winston
Journal:  Hum Reprod       Date:  2002-09       Impact factor: 6.918

5.  Later-onset Fabry disease: an adult variant presenting with the cramp-fasciculation syndrome.

Authors:  Christopher S Nance; Christopher J Klein; Maryam Banikazemi; Steven H Dikman; Robert G Phelps; Justin C McArthur; Moses Rodriguez; Robert J Desnick
Journal:  Arch Neurol       Date:  2006-03

6.  Identity and psychological ownership in chronic illness and disease state.

Authors:  W Karnilowicz
Journal:  Eur J Cancer Care (Engl)       Date:  2010-08-26       Impact factor: 2.520

7.  Consequences of a global enzyme shortage of agalsidase beta in adult Dutch Fabry patients.

Authors:  Bouwien E Smid; Saskia M Rombach; Johannes M F G Aerts; Symen Kuiper; Mina Mirzaian; Hermen S Overkleeft; Ben J H M Poorthuis; Carla E M Hollak; Johanna E M Groener; Gabor E Linthorst
Journal:  Orphanet J Rare Dis       Date:  2011-10-31       Impact factor: 4.123

8.  Patient decisions for disclosure of secondary findings among the first 200 individuals undergoing clinical diagnostic exome sequencing.

Authors:  Layla Shahmirzadi; Elizabeth C Chao; Erika Palmaer; Melissa C Parra; Sha Tang; Kelly D Farwell Gonzalez
Journal:  Genet Med       Date:  2013-10-10       Impact factor: 8.822

Review 9.  Counseling Challenges with Variants of Uncertain Significance and Incidental Findings in Prenatal Genetic Screening and Diagnosis.

Authors:  Lauren Westerfield; Sandra Darilek; Ignatia B van den Veyver
Journal:  J Clin Med       Date:  2014-09-12       Impact factor: 4.241

10.  Alpha-Galactosidase A p.A143T, a non-Fabry disease-causing variant.

Authors:  Malte Lenders; Frank Weidemann; Christine Kurschat; Sima Canaan-Kühl; Thomas Duning; Jörg Stypmann; Boris Schmitz; Stefanie Reiermann; Johannes Krämer; Daniela Blaschke; Christoph Wanner; Stefan-Martin Brand; Eva Brand
Journal:  Orphanet J Rare Dis       Date:  2016-05-04       Impact factor: 4.123

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  2 in total

Review 1.  Patients' views on variants of uncertain significance across indications.

Authors:  Kristin Clift; Sarah Macklin; Colin Halverson; Jennifer B McCormick; Abd Moain Abu Dabrh; Stephanie Hines
Journal:  J Community Genet       Date:  2019-08-20

2.  Cardiomyopathy associated with the Ala143Thr variant of the α-galactosidase A gene.

Authors:  Kati Valtola; Juanita Nino-Quintero; Marja Hedman; Line Lottonen-Raikaslehto; Tomi Laitinen; Maleeha Maria; Ilkka Kantola; Anita Naukkarinen; Markku Laakso; Johanna Kuusisto
Journal:  Heart       Date:  2020-01-16       Impact factor: 5.994

  2 in total

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