A Simple, Universal, and Cost-Efficient Digital PCR Method for the Targeted Analysis of Copy Number Variations.


Journal

Clinical chemistry
ISSN: 1530-8561
Titre abrégé: Clin Chem
Pays: England
ID NLM: 9421549

Informations de publication

Date de publication:
09 2019
Historique:
received: 09 03 2019
accepted: 13 06 2019
pubmed: 12 7 2019
medline: 23 5 2020
entrez: 12 7 2019
Statut: ppublish

Résumé

Rare copy number variations (CNVs) are a major cause of genetic diseases. Simple targeted methods are required for their confirmation and segregation analysis. We developed a simple and universal CNV assay based on digital PCR (dPCR) and universal locked nucleic acid (LNA) hydrolysis probes. We analyzed the mapping of the 90 LNA hydrolysis probes from the Roche Universal ProbeLibrary (UPL). For each CNV, selection of the optimal primers and LNA probe was almost automated; probes were reused across assays and each dPCR assay included the CNV amplicon and a reference amplicon. We assessed the assay performance on 93 small and large CNVs and performed a comparative cost-efficiency analysis. UPL-LNA probes presented nearly 20000000 occurrences on the human genome and were homogeneously distributed with a mean interval of 156 bp. The assay accurately detected all the 93 CNVs, except one (<200 bp), with coefficient of variation <10%. The assay was more cost-efficient than all the other methods. The universal dPCR CNV assay is simple, robust, and cost-efficient because it combines a straightforward design allowed by universal probes and end point PCR, the advantages of a relative quantification of the target to the reference within the same reaction, and the high flexibility of the LNA hydrolysis probes. This method should be a useful tool for genomic medicine, which requires simple methods for the interpretation and segregation analysis of genomic variations.

Sections du résumé

BACKGROUND
Rare copy number variations (CNVs) are a major cause of genetic diseases. Simple targeted methods are required for their confirmation and segregation analysis. We developed a simple and universal CNV assay based on digital PCR (dPCR) and universal locked nucleic acid (LNA) hydrolysis probes.
METHODS
We analyzed the mapping of the 90 LNA hydrolysis probes from the Roche Universal ProbeLibrary (UPL). For each CNV, selection of the optimal primers and LNA probe was almost automated; probes were reused across assays and each dPCR assay included the CNV amplicon and a reference amplicon. We assessed the assay performance on 93 small and large CNVs and performed a comparative cost-efficiency analysis.
RESULTS
UPL-LNA probes presented nearly 20000000 occurrences on the human genome and were homogeneously distributed with a mean interval of 156 bp. The assay accurately detected all the 93 CNVs, except one (<200 bp), with coefficient of variation <10%. The assay was more cost-efficient than all the other methods.
CONCLUSIONS
The universal dPCR CNV assay is simple, robust, and cost-efficient because it combines a straightforward design allowed by universal probes and end point PCR, the advantages of a relative quantification of the target to the reference within the same reaction, and the high flexibility of the LNA hydrolysis probes. This method should be a useful tool for genomic medicine, which requires simple methods for the interpretation and segregation analysis of genomic variations.

Identifiants

pubmed: 31292136
pii: clinchem.2019.304246
doi: 10.1373/clinchem.2019.304246
doi:

Substances chimiques

Oligonucleotides 0
locked nucleic acid 0
DNA 9007-49-2

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1153-1160

Informations de copyright

© 2019 American Association for Clinical Chemistry.

Auteurs

Kévin Cassinari (K)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Olivier Quenez (O)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, F 76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Géraldine Joly-Hélas (G)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Ludivine Beaussire (L)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Nathalie Le Meur (N)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Mathieu Castelain (M)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Alice Goldenberg (A)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Anne-Marie Guerrot (AM)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Anne-Claire Brehin (AC)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Jean-François Deleuze (JF)

Centre National de Recherche en Génomique Humaine, Institut de Génomique, CEA, Evry, France.

Anne Boland (A)

Centre National de Recherche en Génomique Humaine, Institut de Génomique, CEA, Evry, France.

Anne Rovelet-Lecrux (A)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, F 76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Dominique Campion (D)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, F 76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.
Department of Research, Rouvray Psychiatric Hospital, Sotteville-lés-Rouen, France.

Pascale Saugier-Veber (P)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Nicolas Gruchy (N)

Caen University Hospital, Department of Genetics, F 14000, Normandy Center for Genomic and Personalized Medicine, Caen, France.

Thierry Frebourg (T)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Gaël Nicolas (G)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, F 76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Nasrin Sarafan-Vasseur (N)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Pascal Chambon (P)

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics, F76000, Normandy Center for Genomic and Personalized Medicine, Rouen, France; pascal.chambon@chu-rouen.fr.

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