Conventional and Single-Molecule Targeted Sequencing Method for Specific Variant Detection in IKBKG while Bypassing the IKBKGP1 Pseudogene

J Mol Diagn. 2018 Mar;20(2):195-202. doi: 10.1016/j.jmoldx.2017.10.005. Epub 2017 Dec 18.

Abstract

In addition to Sanger sequencing, next-generation sequencing of gene panels and exomes has emerged as a standard diagnostic tool in many laboratories. However, these captures can miss regions, have poor efficiency, or capture pseudogenes, which hamper proper diagnoses. One such example is the primary immunodeficiency-associated gene IKBKG. Its pseudogene IKBKGP1 makes traditional capture methods aspecific. We therefore developed a long-range PCR method to efficiently target IKBKG, as well as two associated genes (IRAK4 and MYD88), while bypassing the IKBKGP1 pseudogene. Sequencing accuracy was evaluated using both conventional short-read technology and a newer long-read, single-molecule sequencer. Different mapping and variant calling options were evaluated in their capability to bypass the pseudogene using both sequencing platforms. Based on these evaluations, we determined a robust diagnostic application for unambiguous sequencing and variant calling in IKBKG, IRAK4, and MYD88. This method allows rapid identification of selected primary immunodeficiency diseases in patients suffering from life-threatening invasive pyogenic bacterial infections.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Bacterial Infections / diagnosis
  • Base Sequence
  • Chromosome Mapping
  • Data Accuracy
  • Diagnosis, Differential
  • Exome
  • Female
  • Genetic Loci
  • High-Throughput Nucleotide Sequencing / methods*
  • Humans
  • I-kappa B Kinase / genetics*
  • Immunologic Deficiency Syndromes / diagnosis
  • Interleukin-1 Receptor-Associated Kinases / genetics
  • Male
  • Myeloid Differentiation Factor 88 / genetics
  • Polymerase Chain Reaction / methods*
  • Polymorphism, Single Nucleotide
  • Pseudogenes / genetics*
  • Sensitivity and Specificity
  • Sequence Analysis, DNA / methods*
  • Structural Homology, Protein

Substances

  • IKBKG protein, human
  • MYD88 protein, human
  • Myeloid Differentiation Factor 88
  • IRAK4 protein, human
  • Interleukin-1 Receptor-Associated Kinases
  • I-kappa B Kinase