Literature DB >> 18607183

What is the clinical utility of DNA testing in patients with familial hypercholesterolaemia?

Steve E Humphries1, Gail Norbury, Sarah Leigh, S Gaye Hadfield, Devikair Nair.   

Abstract

PURPOSE OF REVIEW: Familial hypercholesterolaemia is a common genetic disorder of lipid metabolism in which patients have a significantly elevated risk of early coronary heart disease, which can be substantially lowered by treatment with the statin class of drugs. In many countries in Europe, tracing of relatives using DNA information, once the family mutation has been identified, is being actively carried out. The present review examines the specificity and clinical utility of DNA testing in patients with familial hypercholesterolaemia. RECENT
FINDINGS: Technological progress has improved the detection rate in patients with the strongest clinical suspicion of familial hypercholesterolaemia to more than 70-80%. Patients carrying a mutation have, on average, higher low-density lipoprotein cholesterol levels and greater risk of early coronary heart disease, and studies have reported the utility of DNA information in the identification of affected relatives. More than 1000 different molecular causes of familial hypercholesterolaemia are documented in the University College London database, and although more than 90% of these clearly cause familial hypercholesterolaemia, the remainder require careful interpretation.
SUMMARY: DNA testing, as an adjunct to the measurement of plasma low-density lipoprotein cholesterol levels, has clinical utility in providing an unequivocal diagnosis in patients and in identifying affected relatives at an early age so that they can be offered lifestyle advice and appropriate lipid-lowering therapies. Researchers and DNA diagnostic laboratories need to interpret novel sequence changes with caution in order to avoid a false positive diagnosis.

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Year:  2008        PMID: 18607183     DOI: 10.1097/MOL.0b013e32830636e5

Source DB:  PubMed          Journal:  Curr Opin Lipidol        ISSN: 0957-9672            Impact factor:   4.776


  13 in total

1.  A model of care for familial hypercholesterolaemia: key role for clinical biochemistry.

Authors:  Gerald F Watts; David R Sullivan; Frank M van Bockxmeer; Nicola Poplawski; Ian Hamilton-Craig; Peter M Clifton; Richard C O'Brien; Peter M George; John R Burnett
Journal:  Clin Biochem Rev       Date:  2012-02

Review 2.  Familial Hypercholesterolaemia Diagnosis and Management.

Authors:  Rodrigo Alonso; Leopoldo Perez de Isla; Ovidio Muñiz-Grijalvo; Jose Luis Diaz-Diaz; Pedro Mata
Journal:  Eur Cardiol       Date:  2018-08

3.  Familial Hypercholesterolemia: Cascade Screening in Children and Relatives of the Affected.

Authors:  Nitika Setia; Renu Saxena; J P S Sawhney; Ishwar C Verma
Journal:  Indian J Pediatr       Date:  2018-02-15       Impact factor: 1.967

4.  Cascade Screening for Familial Hypercholesterolemia (FH).

Authors:  Renée M Ned; Eric J G Sijbrands
Journal:  PLoS Curr       Date:  2011-05-23

5.  Molecular spectrum of autosomal dominant hypercholesterolemia in France.

Authors:  Marie Marduel; Alain Carrié; Agnes Sassolas; Martine Devillers; Valérie Carreau; Mathilde Di Filippo; Danièle Erlich; Marianne Abifadel; Alice Marques-Pinheiro; Arnold Munnich; Claudine Junien; Catherine Boileau; Mathilde Varret; Jean-Pierre Rabès
Journal:  Hum Mutat       Date:  2010-11       Impact factor: 4.878

Review 6.  Familial hypercholesterolaemia in children and adolescents: gaining decades of life by optimizing detection and treatment.

Authors:  Albert Wiegman; Samuel S Gidding; Gerald F Watts; M John Chapman; Henry N Ginsberg; Marina Cuchel; Leiv Ose; Maurizio Averna; Catherine Boileau; Jan Borén; Eric Bruckert; Alberico L Catapano; Joep C Defesche; Olivier S Descamps; Robert A Hegele; G Kees Hovingh; Steve E Humphries; Petri T Kovanen; Jan Albert Kuivenhoven; Luis Masana; Børge G Nordestgaard; Päivi Pajukanta; Klaus G Parhofer; Frederick J Raal; Kausik K Ray; Raul D Santos; Anton F H Stalenhoef; Elisabeth Steinhagen-Thiessen; Erik S Stroes; Marja-Riitta Taskinen; Anne Tybjærg-Hansen; Olov Wiklund
Journal:  Eur Heart J       Date:  2015-05-25       Impact factor: 29.983

Review 7.  New Approaches in Detection and Treatment of Familial Hypercholesterolemia.

Authors:  Merel L Hartgers; Kausik K Ray; G Kees Hovingh
Journal:  Curr Cardiol Rep       Date:  2015-12       Impact factor: 2.931

8.  Exploring the complete mutational space of the LDL receptor LA5 domain using molecular dynamics: linking SNPs with disease phenotypes in familial hypercholesterolemia.

Authors:  Vladimir Espinosa Angarica; Modesto Orozco; Javier Sancho
Journal:  Hum Mol Genet       Date:  2016-01-10       Impact factor: 6.150

Review 9.  Genetic Architecture of Familial Hypercholesterolaemia.

Authors:  Mahtab Sharifi; Marta Futema; Devaki Nair; Steve E Humphries
Journal:  Curr Cardiol Rep       Date:  2017-05       Impact factor: 2.931

10.  Whole exome sequencing of familial hypercholesterolaemia patients negative for LDLR/APOB/PCSK9 mutations.

Authors:  Marta Futema; Vincent Plagnol; KaWah Li; Ros A Whittall; H Andrew W Neil; Mary Seed; Stefano Bertolini; Sebastiano Calandra; Olivier S Descamps; Colin A Graham; Robert A Hegele; Fredrik Karpe; Ronen Durst; Eran Leitersdorf; Nicholas Lench; Devaki R Nair; Handrean Soran; Frank M Van Bockxmeer; Steve E Humphries
Journal:  J Med Genet       Date:  2014-07-01       Impact factor: 6.318

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