Improved molecular diagnosis of the common recurrent intragenic deletion mutation in IKBKG in a Filipino family with incontinentia pigmenti

Australas J Dermatol. 2016 May;57(2):150-3. doi: 10.1111/ajd.12407. Epub 2015 Oct 6.

Abstract

Incontinentia pigmenti is a rare, multisystem X-linked dominant genetic disorder caused by mutations in IKBKG, the encoding inhibitor of kappa light polypeptide gene enhancer in B-cells. Almost 80% of all cases result from a recurrent intragenic deletion mutation that removes exon 4-10. At present, this mutation can be detected by a multi-primer polymerase chain reaction (PCR) technique although current protocols may preferentially amplify the wild-type allele and miss the deletion. Here, we report a female infant with incontinentia pigmenti that also affected her mother and sister, and two spontaneously aborted male siblings. We developed a modified PCR amplification method that provides more robust detection of the exon 4-10 deletion mutation, which was demonstrated in all affected females in this pedigree.

Keywords: Blaschko's lines; X-linked; gene deletion; mosaic; vesicle.

Publication types

  • Case Reports

MeSH terms

  • Female
  • Humans
  • I-kappa B Kinase / genetics*
  • Incontinentia Pigmenti / genetics*
  • Infant, Newborn
  • Male
  • Molecular Diagnostic Techniques
  • Pedigree
  • Philippines
  • Polymerase Chain Reaction / methods
  • Sequence Deletion*

Substances

  • IKBKG protein, human
  • I-kappa B Kinase