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

The diagnosis of Wilson disease is not always easy. For many patients, a combination of tests reflecting disturbed copper metabolism may be needed. Testing for ATP7B variants has become part of the routine diagnostic approach. The methods of genetic testing include analysis of the 21 coding exons an...

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Autores principales: Espinós, Carmen, Ferenci, Peter
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7322184/
https://www.ncbi.nlm.nih.gov/pubmed/32613181
http://dx.doi.org/10.1016/j.jhepr.2020.100114
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author Espinós, Carmen
Ferenci, Peter
author_facet Espinós, Carmen
Ferenci, Peter
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description The diagnosis of Wilson disease is not always easy. For many patients, a combination of tests reflecting disturbed copper metabolism may be needed. Testing for ATP7B variants has become part of the routine diagnostic approach. The methods of genetic testing include analysis of the 21 coding exons and intronic flanking sequences, in which exons with recurrent variants would be prioritised depending on the mutation frequency in the local population. If sequencing the entire ATP7B gene cannot identify 2 variants and the suspicion for Wilson disease is high, after reviewing the clinical data, WES (whole-exome sequencing) or WGS (whole-genome sequencing) could be applied. A workflow based on the type and number of ATP7B variants responsible for Wilson disease is proposed. Genetic testing is indicated for confirmation of diagnosis, family screening, and screening of newborns and infants and in unclear cases suspected of suffering from Wilson disease. However, genetic testing is not a routine screening test for Wilson disease. If no additional variants can be identified, it can be assumed that other hereditary disorders may mimic Wilson disease (congenital disorders of glycosylation, MEDNIK syndrome, idiopathic or primary copper toxicoses).
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spelling pubmed-73221842020-06-30 Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice? Espinós, Carmen Ferenci, Peter JHEP Rep Review The diagnosis of Wilson disease is not always easy. For many patients, a combination of tests reflecting disturbed copper metabolism may be needed. Testing for ATP7B variants has become part of the routine diagnostic approach. The methods of genetic testing include analysis of the 21 coding exons and intronic flanking sequences, in which exons with recurrent variants would be prioritised depending on the mutation frequency in the local population. If sequencing the entire ATP7B gene cannot identify 2 variants and the suspicion for Wilson disease is high, after reviewing the clinical data, WES (whole-exome sequencing) or WGS (whole-genome sequencing) could be applied. A workflow based on the type and number of ATP7B variants responsible for Wilson disease is proposed. Genetic testing is indicated for confirmation of diagnosis, family screening, and screening of newborns and infants and in unclear cases suspected of suffering from Wilson disease. However, genetic testing is not a routine screening test for Wilson disease. If no additional variants can be identified, it can be assumed that other hereditary disorders may mimic Wilson disease (congenital disorders of glycosylation, MEDNIK syndrome, idiopathic or primary copper toxicoses). Elsevier 2020-04-18 /pmc/articles/PMC7322184/ /pubmed/32613181 http://dx.doi.org/10.1016/j.jhepr.2020.100114 Text en © 2020 The Authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Review
Espinós, Carmen
Ferenci, Peter
Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title_full Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title_fullStr Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title_full_unstemmed Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title_short Are the new genetic tools for diagnosis of Wilson disease helpful in clinical practice?
title_sort are the new genetic tools for diagnosis of wilson disease helpful in clinical practice?
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7322184/
https://www.ncbi.nlm.nih.gov/pubmed/32613181
http://dx.doi.org/10.1016/j.jhepr.2020.100114
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