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Short amplicon reverse transcription-polymerase chain reaction detects aberrant splicing in genes with low expression in blood missed by ribonucleic acid sequencing analysis for clinical diagnosis.
Wai, Htoo A; Constable, Matthew; Drewes, Cosima; Davies, Ian C; Svobodova, Eliska; Dempsey, Esther; Saggar, Anand; Homfray, Tessa; Mansour, Sahar; Douzgou, Sofia; Barr, Kate; Mercer, Catherine; Hunt, David; Douglas, Andrew G L; Baralle, Diana.
Afiliación
  • Wai HA; Human Development and Health, Faculty of Medicine, Southampton General Hospital, University of Southampton, Southampton, UK.
  • Constable M; Human Development and Health, Faculty of Medicine, Southampton General Hospital, University of Southampton, Southampton, UK.
  • Drewes C; Human Development and Health, Faculty of Medicine, Southampton General Hospital, University of Southampton, Southampton, UK.
  • Davies IC; Human Development and Health, Faculty of Medicine, Southampton General Hospital, University of Southampton, Southampton, UK.
  • Svobodova E; Department of Experimental Biology, Faculty of Science, Masaryk University, Brno, Czech Republic.
  • Dempsey E; Department of Clinical Genetics, St George's University of London, London, UK.
  • Saggar A; Department of Clinical Genetics, St George's University of London, London, UK.
  • Homfray T; Department of Clinical Genetics, St George's University of London, London, UK.
  • Mansour S; Department of Clinical Genetics, St George's University of London, London, UK.
  • Douzgou S; Manchester Centre for Genomic Medicine, St Mary's Hospital, Manchester University NHS Foundation Trust, Health Innovation Manchester, Manchester, UK.
  • Barr K; Division of Evolution and Genomic Sciences, Faculty of Biology, Medicine and Health, School of Biological Sciences, University of Manchester, Manchester, UK.
  • Mercer C; Bristol Regional Clinical Genetics Service, St Michael's Hospital, Bristol, UK.
  • Hunt D; Department of Experimental Biology, Faculty of Science, Masaryk University, Brno, Czech Republic.
  • Douglas AGL; Human Development and Health, Faculty of Medicine, Southampton General Hospital, University of Southampton, Southampton, UK.
  • Baralle D; Wessex Clinical Genetics Service, University Hospital Southampton NHS Foundation Trust, Southampton, UK.
Hum Mutat ; 43(7): 963-970, 2022 07.
Article en En | MEDLINE | ID: mdl-35476365
Use of blood RNA sequencing (RNA-seq) as a splicing analysis tool for clinical interpretation of variants of uncertain significance (VUSs) found via whole-genome and exome sequencing can be difficult for genes that have low expression in the blood due to insufficient read count coverage aligned to specific genes of interest. Here, we present a short amplicon reverse transcription-polymerase chain reaction(RT-PCR) for the detection of genes with low blood expression. Short amplicon RT-PCR, is designed to span three exons where an exon harboring a variant is flanked by one upstream and one downstream exon. We tested short amplicon RT-PCRs for genes that have median transcripts per million (TPM) values less than one according to the genotype-tissue expression database. Median TPM values of genes analyzed in this study are SYN1 = 0.8549, COL1A1 = 0.6275, TCF4 = 0.4009, DSP = .2894, TTN = 0.2851, COL5A2 = 0.1036, TERT = 0.04452, NTRK2 = 0.0344, ABCA4 = 0.00744, PRPH = 0, and WT1 = 0. All these genes show insufficient exon-spanning read coverage in our RNA-seq data to allow splicing analysis. We successfully detected all genes tested except PRPH and WT1. Aberrant splicing was detected in SYN1, TCF4, NTRK2, TTN, and TERT VUSs. Therefore, our results show short amplicon RT-PCR is a useful alternative for the analysis of splicing events in genes with low TPM in blood RNA for clinical diagnostics.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: ARN / Empalme Alternativo Tipo de estudio: Diagnostic_studies Límite: Humans Idioma: En Revista: Hum Mutat Asunto de la revista: GENETICA MEDICA Año: 2022 Tipo del documento: Article Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: ARN / Empalme Alternativo Tipo de estudio: Diagnostic_studies Límite: Humans Idioma: En Revista: Hum Mutat Asunto de la revista: GENETICA MEDICA Año: 2022 Tipo del documento: Article Pais de publicación: Estados Unidos