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Published on: April 21, 2017
[A Case of Pediatric C1 Dysplasia with Ruptured Aneurysm in Collateral Plexiform Arterial Network]
Akihiro Mizuno1, Kenichi Haraguchi, Takeshi Okada
1Department of Neurosurgery, Kainan Hospital.
Insights
A rare C1 dysplasia caused a brain aneurysm in a child, treated with coil embolization. The aneurysm recurred due to hemodynamic factors, highlighting the vulnerability of collateral networks.
Area of Science:
- Neurology
- Vascular Surgery
- Pediatric Neurosurgery
Background:
- Subarachnoid hemorrhage in children is rare and often associated with vascular malformations.
- Internal carotid artery (ICA) dysplasia, particularly at the C1 segment, is an uncommon cause of intracranial aneurysms.
Purpose of the Study:
- To report a case of pediatric C1 dysplasia presenting as a saccular aneurysm.
- To discuss the diagnostic and treatment challenges of this rare condition.
- To explore the potential mechanisms of aneurysm recurrence.
Main Methods:
- Case presentation of a 3-year-old girl with subarachnoid hemorrhage.
- Diagnostic imaging including Head CT and digital subtraction angiography (DSA).
- Interventional treatment with coil embolization and follow-up assessment.
Main Results:
- DSA revealed right ICA C1 segment dysplasia with a collateral plexiform arterial network forming a saccular aneurysm.
- Successful initial coil embolization resulted in no neurological deficit.
- Recurrence of the aneurysm was observed four months post-procedure.
Conclusions:
- C1 dysplasia can lead to complex intracranial aneurysms in pediatric patients.
- Hemodynamic forces and the nature of collateral networks may contribute to aneurysm recurrence after embolization.
- Further research into the pathophysiology and long-term management of these rare aneurysms is warranted.
Abstract:
A 3-year-old girl was admitted to our hospital with symptoms including headache, nausea, and vomiting. Head CT scan showed subarachnoid hemorrhage in the right carotid cistern. Digital subtraction angiography revealed right internal caortid artery(ICA)malformation at the C1 segment with collateral plexiform arterial network. The right ICA branched into posterior communicating artery and anterior choroidal artery(AChoA)and the ICA was decreased in caliber. The distal portion of the C1 segment of the ICA continued to the collateral plexiform arterial network, forming a saccular aneurysm. The plexiform arterial network connected to the right AChoA and the anterior communicating artery and continued to the distal portion of the right M1 segment. Right cervical carotid artery was normal. There was no transdural collateral flow from the right external carotid artery. Genetic analysis of a variant of RING finger protein 213 was negative. We diagnosed this patient with C1 dysplasia. We performed coil embolization for the aneurysm. The patient was discharged without any neurological deficit. Four months after the surgery, recurrence of the aneurysm was observed. We suspected that the aneurysm was formed due to hemodynamic mechanism and vulnerability of the collateral plexiform arterial network.
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