Grave aortic aneurysmal dilatation in DOCK8 deficiency

Mod Rheumatol. 2014 Jul;24(4):690-3. doi: 10.3109/14397595.2013.874735. Epub 2014 Feb 11.

Abstract

Hyperimmunoglobulin E syndrome (HIES) is a primary immunodeficiency disorder with multisystem abnormalities including the vascular system. We report a child with autosomal recessive (AR)-HIES secondary to dedicator of cytokinesis 8 (DOCK8) deficiency who developed critical aortic aneurysm involving the ascending aorta and aortic arch with narrowing of descending aorta that was successfully managed surgically. This report highlights the underrecognized and serious complication of DOCK8 deficiency that could contribute to significant morbidity and mortality in such patients.

Keywords: Aneurysm; Aorta; DOCK8; Hyperimmunoglobulin E syndrome; Vasculitis.

Publication types

  • Case Reports

MeSH terms

  • Aortic Aneurysm / complications*
  • Aortic Aneurysm / surgery
  • Child
  • Guanine Nucleotide Exchange Factors / deficiency*
  • Humans
  • Job Syndrome / complications*
  • Male
  • Treatment Outcome

Substances

  • DOCK8 protein, human
  • Guanine Nucleotide Exchange Factors