Aneurysm on a persistent hypoglossal artery

Summary The authors report a patient with saccular aneurysm on a left persistent hypoglossal artery who suffered subarachnoid haemorrhage from an aneurysm of the right distal anterior cerebral artery. A review of the literature emphasizes the rarity of the occurrence of an aneurysm on a persistent primitive hypoglossal artery itself.     

A case report: persistent primitive hypoglossal artery aneurysm

The authors describe a case of persistent primitive hypoglossal artery aneurysm in a 42-year-old woman who had complained of headache, mainly in the occiput, for 5 days prior to admission. Because of a sudden exacerbation of the headache associated with vomiting, she was hospitalized on July 31, 1988. On admission, a cranial computed tomography scan demonstrated a high density lesion in the basal cisterns which suggested subarachnoid hemorrhage (SAH). Right carotid angiography revealed a persistent primitive hypoglossal artery and an aneurysm arising from this artery at the junction of the posterior inferior cerebellar artery. Bilateral vertebral arteries were shown to be hypoplastic. This was followed by a right suboccipital craniectomy on the 6th hospital day at which time a neck clipping was made. Her postoperative course was uneventful. On discharge on August 22, she was ambulatory and had no neurological deficit except for a mild hoarseness which developed after surgery. Well over one hundred cases of persistent primitive hypoglossal artery aneurysm have been reported. However, as far as we could discern, there have been only 9 cases of persistent primitive hypoglossal artery aneurysm including this present case. Most of the cases had SAH due to the rupture of these aneurysms with favorable outcome after the surgery. In addition, some embryological considerations were made.     

Basilar bifurcation aneurysms associated with persistent primitive hypoglossal artery

We report two cases of ruptured basilar bifurcation aneurysm associated with a persistent primitive hypoglossal artery. Angiograms revealed low-positioned aneurysms; in both cases bilateral vertebral arteries and posterior communicating arteries were hypoplastic or aplastic. Both aneurysms were successfully clipped via subtemporal transtentorial approach through the craniotomy ipsilateral to the side of the primitive hypoglossal artery. The ipsilateral craniotomy and exposure of the cervical carotid artery were helpful for obtaining the proximal control of the basilar artery needed to perform the clipping procedure with safety.     

Posterior inferior cerebellar artery aneurysm associated with persistent primitive hypoglossal artery

A 49-year-old woman presented with subarachnoid hemorrhage (SAH) from an aneurysm associated with a persistent primitive hypoglossal artery (PPHA) manifesting as sudden onset of headache, but without neurological deficits. Conventional computed tomography (CT) of her head showed no abnormality but lumbar tap indicated SAH. Three-dimensional (3D)-CT angiography showed a PPHA originating from the internal carotid artery and an aneurysm of the posterior inferior cerebellar artery at the junction with the remnant hypoplastic vertebral artery. 3D-CT angiography was essentially useful for presurgical planning to determine the extent of craniotomy and the space for possible temporary clipping, and confirmed the diagnosis of aneurysmal SAH. The aneurysm was clipped and she returned to her job 4 weeks later. Cerebral angiography is the golden standard technique to diagnose PPHA, but 3D-CT angiography can be recommended for presurgical evaluation, especially in patients with complex and anomalous anatomical structures.     

A large aneurysm of the persistent primitive hypoglossal artery.

An aneurysm of a persistent primitive hypoglossal artery (PHA) particularly at its junction with the basilar artery, often poses therapeutic problems. This is attributable not only to the size and location of the aneurysm but also to the fact that the persistent PHA is functionally the single artery providing blood in posterior circulation. We report a 31-year-old man with a large aneurysm of the persistent PHA at its junction with the basilar artery and review the existing literature. We clipped the broad neck of the aneurysm through a lateral suboccipital craniectomy; however, the patient suffered a second episode of subarachnoid hemorrhage. Angiography disclosed a slipped clip incompletely obstructing the persistent PHA. He received no further treatment for the aneurysm because of his deteriorating condition. Direct surgical treatment, although it failed to obliterate the aneurysm neck in our patient, should be recommended for a persistent PHA aneurysm.     

Moyamoya disease with persistent primitive hypoglossal artery. Case report

A 44-year-old female developed aphasia and visual disturbance of the left eye. Four-vessel angiography showed occlusion of the extra- and intracranial portions of the right internal carotid artery, severe stenosis of the left internal carotid fork, bilateral moyamoya vessels, and a persistent primitive hypoglossal artery on the left side. Measurement of cerebral blood flow by 133xe inhalation revealed decreased flow and no response to acetazolamide loading in the left cerebral hemisphere. The patient underwent superficial temporal artery-middle cerebral artery anastomosis, first on the left and then on the right side. There have been no other reported cases of moyamoya disease associated with a persistent primitive hypoglossal artery. Although the cause of moyamoya disease remains to be established, it is generally believed that stenotic changes of the carotid fork cause the development of moyamoya vessels as collateral pathways. According to this theory, there is no significant etiological relationship between moyamoya disease and persistent primitive hypoglossal artery. However, persistent primitive hypoglossal artery is compatible with moyamoya disease in terms of providing a collateral pathway via the vertebrobasilar system.     

Ruptured persistent primitive hypoglossal artery aneurysm: case report]

Persistent primitive hypoglossal artery is less common than persistent primitive trigeminal artery. About one hundred examples of such hypoglossal arteries have been demonstrated by angiography. The origin of persistent primitive hypoglossal artery is the cervical segment of the internal carotid artery, usually at the level of the first to second cervical vertebrae. The artery then enters, with varying degrees of tortuosity, the anterior condyloid (hypoglossal) canal and joints the basilar artery immediately above its lower end. When enlargement of this canal is identified, the presence of a persistent primitive hypoglossal artery should be strongly suspected. The homolateral vertebral artery is frequently hypoplasia. A 66-year-old man was brought to our hospital due to faintness. CT showed thick subarachnoid hemorrhage. Angiography showed that a persistent primitive hypoglossal artery aneurysm was present, but the posterior communicating artery was absent. Right vertebral angiography showed extravasation due to re-rupture of the aneurysm. An operation was performed at day 0 using the left transcondylar approach despite deterioration of SAH grading. Intraoperative re-rupturing occurred and the lower cranial nerves clustered around the aneurysm, so the aneurysm was partially clipped on the dome. The second angiography was carried out at day 10, and there was no vasospasm. Palsy of the lower cranial nerves appeared transiently. A ventricle-peritoneum shunt was required due to normal pressure hydrocephalus, but the patient was discharged with no neurological deficits.     

Rare variant of persistent primitive hypoglossal artery, arising from the external carotid artery

A 63-year-old man presented with an extremely rare variant of persistent primitive hypoglossal artery (PHA), which was found incidentally during examination for a contralateral asymptomatic internal carotid artery (ICA) stenosis. This anastomotic vessel arose from the external carotid artery, not the ICA, and joined the vertebrobasilar artery through the hypoglossal canal. Persistent PHA is rare and the reported incidence is 0.027-0.26%. Recognition of the existence of this variant vessel and preservation during neuroradiologic intervention or surgery is important to prevent possible ischemic complications.     

An autopsy case of persistent primitive hypoglossal artery with multiple cerebral aneurysms

An autopsy case of persistent primitive hypoglossal artery (PPHA) with multiple cerebral aneurysms is reported. A 54-year-old man with subarachnoid hemorrhage was admitted to Kuwana Hospital three days after the onset. The patient was stuporous and had stiffness of the neck. A computed tomogram showed hematoma in the interhemispheric fissure, subarachnoid hemorrhage in the basal cisterns and bilateral Sylvian fissures, and maxked dilatation of ventricles. Cerebral angiogram revealed the left PPHA and multiple aneurysms at the right anterior cerebral artery (A 2) (ruptured), anterior communicating artery, left anterior cerebral artery (A 1), left internal carotid-anterior choroidal artery junction, right internal carotid artery (C 1), and right middle cerebral artery. Neck clipping of the ruptured aneurysm and ventricular drainage were performed on the day of admission. Eight days after admission he died of rupture of the residual aneurysm. In pathological study, the PPHA was originated from the extracranial portion of the left internal carotid artery, 2 cm distal from the cervical carotid bifurcation, entered the intracranial space through the hypoglossal foramen, and turned into the basilar artery. There were six aneurysms which were shown on cerebral angiogram and another aneurysm on the left anterior inferior cerebellar artery. Microscopic examination revealed atherosclerotic change of the PPHA, true aneurysmal changes of the seven aneurysms and defect of tunica media (Forbus' medial gap) at all of the arterial bifurcations without early aneurysmal changes.     

Endovascular treatment of AVM-associated aneurysm of anterior inferior cerebellar artery through persistent primitive hypoglossal artery

We present a patient with the combination of persistent primitive hypoglossal artery and the origin of left common carotid artery from the brachiocephalic trunk, who had subarachnoid haemorrhage caused by a ruptured aneurysm. The aneurysm was on distal anterior inferior cerebellar artery which was a feeder to cerebellar arterio-venous malformation.     

Persistent primitive hypoglossal artery aneurysm--case report

The aneurysm arising from a persistent primitive hypoglossal artery (PHA) is rare, and only 13 such cases have been reported in literature. We present a 62-year-old woman with an aneurysm of PHA at its junction with the basilar artery. The patient consulted our hospital for a transient loss of consciousness and headaches. No neurological deficit was found, but MRI and MRA showed an aneurysm of the vertebrobasilar junction. Cerebral angiogram after admission showed the aneurysm of PHA at its junction with the basilar artery. Perspective 3D-CTA and 3D-T2 weighted MR images were composed to simulate the condition and aneurysmal surgery via the transcondylar approach was carried out. The aneurysm was successfully clipped and the patient was discharged with no neurological deficits. Perspective 3 D-CTA and MRI simulation were very useful for this operation.     

Endarterectomy for persistent primitive hypoglossal artery--case report.

Persistent primitive hypoglossal artery, an anastomosis between the carotid artery and the vertebrobasilar system, is found in about 0.05% of cerebral angiograms. Though usually asymptomatic, it may occasionally cause ischemic disease. A 62-year-old male presented with left hemiparesis. Right carotid angiograms demonstrated a primitive hypoglossal artery originating from the internal carotid artery at the 2nd cervical spine. This artery supplied almost all blood to the basilar artery system. A marked stenosis extended from the origin of the internal carotid artery to the primitive hypoglossal artery. An endarterectomy of the internal carotid and primitive hypoglossal arteries was performed using a special internal shunt 46 days after the onset. Sudden arterial bleeding from the incised part of the internal carotid artery occurred 12 days after the operation. The carotid artery was resutured. The rupture of the carotid artery appeared to be caused by an infection of Pseudomonas aeruginosa, which was detected by culture of the chronic ear discharge. Rupture of the vessel wall due to infection is an important complication after endarterectomy. This is the second reported endarterectomy of the primitive hypoglossal artery.     

Ruptured aneurysm of a persistent primitive hypoglossal artery treated by endovascular approach--case report and literature review

BACKGROUND: A persistent PHA is the second most common of the embryonic carotid-basilar anastomoses that fail to regress in the embryo. The fact that PHA often is functionally a single artery providing blood to the posterior circulation poses challenging therapeutic problems in case of an aneurysm located on the PHA. CASE DESCRIPTION: A 46-year-old woman presented with SAH due to a large ruptured aneurysm of the left PHA. Identification of such an artery by CT angiogram is the proposed cold standard. The aneurysm was obliterated by coil embolization. To our knowledge, this is the first reported case of aneurysm located on a primitive persistent hypoglossal artery that was endovascularly treated. CONCLUSION: Aneurysms located on a persistent PHA can be treated safely and effectively via an endovascular approach.     

Aneurysm of a persistent sciatic artery

We report a case of an aneurysm of a persistent sciatic artery which caused buttock pain. Preoperative diagnosis is very difficult. However, awareness of the presence of this rare embryonic abnormality is important, especially in elderly persons with atherosclerotic changes. Sagittal magnetic resonance imaging (MRI) was very useful in reaching this diagnosis. Received: 15 February 1994     

Persistent primitive hypoglossal artery aneurysms--report of two cases.

The authors present two patients with subarachnoid hemorrhage caused by ruptured intracranial saccular aneurysms of the persistent primitive hypoglossal artery. A standard unilateral suboccipital approach in one patient resulted in incomplete neck clipping since the operative field was restricted by a protruding jugular tubercle. Successful aneurysmal neck clipping was achieved in the second patient via a unilateral-transcondylar-suboccipital approach with resection of the jugular tubercle and rim of the foramen magnum.     

Large arteriovenous malformation associated with persistent primitive hypoglossal artery--case report.

A rare case of large arteriovenous malformation (AVM) with persistent primitive hypoglossal artery in a 43-year-old male was treated by staged embolization, followed by total removal. AVM associated with carotid-basilar anastomosis is quite rare, but the incidence of AVM in patients with carotid-basilar anastomosis is high. AVM associated with persistent carotid-basilar anastomosis has no distinguishing features compared with ordinary AVM, but participation of the posterior circulation as a feeder is characteristic.     

Persistent primitive hypoglossal artery associated with proximal posterior inferior cerebellar artery aneurysm

BACKGROUND: A persistent primitive hypoglossal artery (PPHA) is a rare anomaly. The association of PPHA with intracranial aneurysms of the artery has also been rarely reported. We surgically treated a case of PPHA associated with a ruptured saccular aneurysm at the proximal posterior inferior cerebellar artery (PICA). CASE DESCRIPTION: The patient was admitted because of subarachnoid hemorrhaging. Angiography and three-dimensional computed tomography (CT) angiography (3D-CTA) demonstrated a left PPHA entering the posterior fossa through the left large hypoglossal canal. The left vertebral artery was absent. A saccular aneurysm was found at the junction of the PPHA and the proximal PICA. 3D-CTA showed not only the aneurysm itself but also the anatomical relationship between the aneurysm and the surrounding structures. Therefore, 3D-CTA was very useful in planning the surgery. The neck of the aneurysm was clipped through a far lateral approach associated with a C1 laminectomy, because this case had a large posterior condylar emissary vein and the aneurysm was located just posteroinferior to the hypoglossal canal. CONCLUSIONS: A case of PPHA associated with an aneurysm at the proximal PICA is reported. This case not only had a large hypoglossal canal but also had a huge posterior condylar emissary vein in the large posterior condylar canal. Anomalous structures associated with PPHA are also discussed. Finally, 3D-CTA proved to be very useful in planning the optimal surgical modality around the lateral portion of the foramen magnum.