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Persistent primitive hypoglossal artery associated with arteriovenous malformation--case report

K Yamanaka1, K Noguchi, K Hayasaki

  • 1Department of Neurosurgery, Hayashi Hospital, Osaka, Japan.

Neurologia Medico-Chirurgica
|November 1, 1990
PubMed

Insights

A rare case of persistent primitive hypoglossal artery (PPHA) associated with an arteriovenous malformation (AVM) was successfully treated with complete surgical removal. This case highlights a rare but treatable condition involving vascular anomalies.

Area of Science:

  • Neurology
  • Neurosurgery
  • Vascular Medicine

Background:

  • Persistent primitive hypoglossal artery (PPHA) is a rare congenital vascular anomaly.
  • Intracranial arteriovenous malformations (AVMs) are complex vascular lesions that can lead to hemorrhage.
  • The co-occurrence of PPHA and intracranial AVM is exceptionally uncommon.

Observation:

  • A 46-year-old male presented with sudden severe headache and transient unconsciousness, indicative of subarachnoid hemorrhage.
  • Computed tomography and angiography revealed a left cerebellar hemisphere AVM associated with an ipsilateral PPHA.
  • This represents the fourth reported case of PPHA associated with an intracranial AVM.

Findings:

  • The patient underwent complete surgical resection of the arteriovenous malformation.
  • The patient recovered without neurological deficit and was discharged within one month.
  • This is the first reported case of PPHA associated with AVM treated by total AVM removal.

Implications:

  • The successful surgical management of this rare PPHA-AVM complex suggests that complete resection is a viable treatment option.
  • This case contributes to the limited literature on PPHA-AVM, emphasizing its rarity (3.0% of reported PPHA cases).
  • Further research into the embryological basis and optimal management strategies for PPHA-AVM is warranted.

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