Intracranial Aneurysms: Pathology, Genetics, and Molecular Mechanisms

Neuromolecular Med. 2019 Dec;21(4):325-343. doi: 10.1007/s12017-019-08537-7. Epub 2019 May 4.

Abstract

Intracranial aneurysms (IA) are local dilatations in cerebral arteries that predominantly affect the circle of Willis. Occurring in approximately 2-5% of adults, these weakened areas are susceptible to rupture, leading to subarachnoid hemorrhage (SAH), a type of hemorrhagic stroke. Due to its early age of onset and poor prognosis, SAH accounts for > 25% of years lost for all stroke victims under the age of 65. In this review, we describe the cerebrovascular pathology associated with intracranial aneurysms. To understand IA genetics, we summarize syndromes with elevated incidence, genome-wide association studies (GWAS), whole exome studies on IA-affected families, and recent research that established definitive roles for Thsd1 (Thrombospondin Type 1 Domain Containing Protein 1) and Sox17 (SRY-box 17) in IA using genetically engineered mouse models. Lastly, we discuss the underlying molecular mechanisms of IA, including defects in vascular endothelial and smooth muscle cells caused by dysfunction in mechanotransduction, Thsd1/FAK (Focal Adhesion Kinase) signaling, and the Transforming Growth Factor β (TGF-β) pathway. As illustrated by THSD1 research, cell adhesion may play a significant role in IA.

Keywords: Animal models; Etiology; Genetics; Intracranial aneurysm; Subarachnoid hemorrhage; THSD1.

Publication types

  • Research Support, N.I.H., Extramural
  • Review

MeSH terms

  • Aneurysm, Ruptured / complications
  • Animals
  • Arteritis / complications
  • Arteritis / pathology
  • Case-Control Studies
  • Cerebral Arteries / ultrastructure
  • Disease Models, Animal
  • Endothelial Cells / metabolism
  • Endothelial Cells / pathology
  • Exome Sequencing
  • Focal Adhesions
  • Genetic Predisposition to Disease
  • Genome-Wide Association Study
  • Hemorheology
  • Humans
  • Incidence
  • Intracranial Aneurysm* / complications
  • Intracranial Aneurysm* / epidemiology
  • Intracranial Aneurysm* / genetics
  • Intracranial Aneurysm* / pathology
  • Mammals
  • Mechanotransduction, Cellular
  • Mice
  • Myocytes, Smooth Muscle / pathology
  • SOXF Transcription Factors / physiology
  • Subarachnoid Hemorrhage / etiology
  • Syndrome
  • Thrombospondins / physiology
  • Transforming Growth Factor beta / physiology
  • Zebrafish

Substances

  • SOX17 protein, human
  • SOXF Transcription Factors
  • THSD1 protein, human
  • Thrombospondins
  • Transforming Growth Factor beta