Implementing long-read sequencing

  • Research type

    Research Study

  • Full title

    Implementing long-read sequencing in a clinical service costs and benefits

  • IRAS ID

    346352

  • Contact name

    Natalie Chandler

  • Contact email

    Natalie.chandler@gosh.nhs.uk

  • Sponsor organisation

    Great Ormond Street Hospital for Children NHS Foundation Trust

  • Duration of Study in the UK

    2 years, 6 months, 22 days

  • Research summary

    Next-generation sequencing (NGS) is a powerful molecular tool that is routinely used in diagnostic genetics laboratories worldwide. NGS enables sequencing of an individual’s genome in short DNA fragments which are computationally organised against a ‘normal’ reference genome. This technology, referred to as ‘short-read’ NGS is effective at sequencing much of the human genome, however gaps can arise due to the complex and repetitive nature of some genomic regions. Crucially, disease-causing variations hidden in these gaps may result in a missed diagnosis. Alternative sequencing technologies, referred to as ‘long-read’, significantly increase the size of the sequenced fragments, thereby reducing the number of gaps, and potentially increasing the diagnostic yield.

    The overarching aim is to demonstrate how long-read NGS can overcome issues encountered by short-read sequencing, with a particular focus on specific genes and genetic disorders that are tested for at GOSH. Furthermore, the possible economic impact of long-read sequencing will be investigated to determine if this approach could result in a higher diagnostic yield at a comparable or cheaper cost to the NHS than the current diagnostic methodologies. Long-read sequencing has the potential to transform the clinical diagnostic landscape of unsolved cases.

  • REC name

    North West - Liverpool Central Research Ethics Committee

  • REC reference

    25/NW/0089

  • Date of REC Opinion

    21 Mar 2025

  • REC opinion

    Favourable Opinion