Neonatal Aneurysm Rupture in a Child with a De Novo Variant to ANKRD17.
Rebecca SilversteinMichael KuwabaraBrian L AppavuPublished in: Child neurology open (2022)
Ankyrin repeat domain 17 (ANKRD17) is postulated to play a role in the integrity of blood vessels and has been reported to be associated with developmental delays, epilepsy, and growth restriction. Whereas ANKRD17-deficient mice have been demonstrated to experience catastrophic hemorrhages, vascular malformations have not been reported in human patients with pathogenic variants to ANKRD17. We report a term male neonate with a heterozygous de novo variant to ANKRD17 (ANKRD17; c6988 C > G, P.[P2330a]) who experienced subarachnoid hemorrhage from a ruptured aneurysm involving the left middle cerebral artery. He experienced acute symptomatic seizures and required clipping of his aneurysm at 35 days of life, later progressing to developing multifocal drug-resistant epilepsy. To our knowledge, this case represents the first report of a cerebrovascular malformation from a patient with ANKRD17. Further work is needed to investigate whether pathogenic variants to ANKRD17 can lead to cerebral aneurysms or other cerebrovascular malformations in children.
Keyphrases
- subarachnoid hemorrhage
- drug resistant
- middle cerebral artery
- brain injury
- coronary artery
- multidrug resistant
- cerebral ischemia
- abdominal aortic aneurysm
- endothelial cells
- healthcare
- young adults
- early onset
- copy number
- liver failure
- mental health
- case report
- intensive care unit
- internal carotid artery
- pseudomonas aeruginosa
- genome wide
- drug induced
- extracorporeal membrane oxygenation
- respiratory failure
- temporal lobe epilepsy