ABSTRACT
Persistent primitive hypoglossal artery (PPHA) is a rare embryological carotid-basilar anastomosis. Diagnosis is by imaging and computed tomography angiography is the most common methods employed. Herein we present a case of a 51 years-old Indian male, who admitted with midnight nonwitnessed syncope. CT angiogram of the brain revealed a very rare caroto-basilar anastomoses anomaly with PPHA.
ABSTRACT
Persistent primitive hypoglossal artery is a relatively rare anatomical variation and a type of persistent carotid-basilar anastomosis. Acute internal carotid artery occlusion associated with persistent primitive hypoglossal artery is rare, and atherothrombotic occlusion is extremely rare. We present a case of acute atherothrombotic internal carotid artery occlusion associated with persistent primitive hypoglossal artery that was successfully treated by endovascular treatment. A 70-year-old male with a history of left internal carotid artery stenosis was transferred to our hospital by ambulance because of abnormal behaviors and aphasia. He was diagnosed with cerebral infarction and left internal carotid artery occlusion. Left carotid angiography revealed the persistent primitive hypoglossal artery arising from the cervical internal carotid artery and complete internal carotid artery occlusion distal to the origin of the persistent primitive hypoglossal artery. Therefore, we performed endovascular treatment. Mechanical thrombectomy was performed under minimal flow arrest with consideration of brain ischemia causing coma. After additional balloon angioplasty, recanalization was achieved, and the patient's symptoms improved. During the 1.5-year follow-up period, no recurrence or restenosis was observed. This report provides evidence that atherosclerotic internal carotid artery stenosis associated with persistent primitive hypoglossal artery can occur even distal to the origin of the persistent primitive hypoglossal artery and that the lesion may become acutely occluded, leading to acute stroke. Endovascular treatment considering brain ischemia was effective in this case.
Subject(s)
Brain Ischemia , Carotid Artery Diseases , Carotid Stenosis , Male , Humans , Aged , Carotid Artery, Internal/diagnostic imaging , Carotid Artery, Internal/surgery , Carotid Stenosis/complications , Carotid Stenosis/diagnostic imaging , Carotid Stenosis/surgery , Carotid Artery Diseases/complications , Carotid Arteries/diagnostic imaging , Carotid Arteries/surgery , Brain Ischemia/complicationsABSTRACT
Persistent primitive hypoglossal artery (PPHA) is a highly uncommon abnormal connection between the internal carotid artery (ICA) and vertebral artery (VA), with reported incidences ranging from 0.027 to 0.26%. Attempting endovascular intervention in such cases presents a considerable challenge as it carries a higher risk of embolization and other procedure-related complications that may affect a wide area of the brain. We present a case study involving the utilization of mechanical thrombectomy (MT) to treat an ischemic stroke in the M1 segment of the middle cerebral artery (MCA) despite the presence of PPHA. Performing mechanical thrombectomy in an anomalous vascular connection is feasible; however, it necessitates heightened vigilance, thorough knowledge of the anatomy, and utmost caution.
ABSTRACT
Background: Persistent primitive hypoglossal artery (PPHA) is a rare residual arterial anastomosis. We placed a CASPER stent using Spider FX as an embolic protection device (EPD) in a patient with internal carotid artery (ICA) stenosis and PPHA. There are no reports of carotid artery stenting (CAS) using a CASPER stent for ICA stenosis with PPHA. We report the EPD strategy used in this case and the usefulness and precautions of CASPER stent insertion for cervical ICA stenosis in association with PPHA. Methods: A 9Fr sheath was placed in the right femoral artery and a 9Fr Branchor balloon guide catheter was guided to the common carotid artery. A Spider FX was placed proximal to the bifurcation of the ICA and the PPHA. A 10 mm × 20 mm CASPER stent was deployed at the site of the stricture with no postoperative ischemic complications. Results: There was no intra-stent occlusion, stenosis, or plaque protrusion immediately after surgery, and no postoperative ischemic complications were observed. Conclusion: CASPER stent deployment with the Spider FX in the ICA and PPHA bifurcation can be considered to be an effective treatment method for ICA stenosis associated with PPHA. However, care should be taken in selecting the appropriate EPDs and stents depending on the location of the stenosis and bifurcation of the PPHA.
ABSTRACT
BACKGROUND: We report a case of symptomatic, progressive stenosis of a persistent primitive hypoglossal artery (PPHA), which was successfully treated with percutaneous transluminal angioplasty (PTA) of the origin of the PPHA. The PPHA is a type of carotid-basilar anastomosis with an incidence of 0.02% to 0.10%. It originates from the internal carotid artery (ICA), passes through the hypoglossal canal, and merges with the basilar artery. In many cases, the ipsilateral vertebral artery is hypoplastic; therefore, PPHA stenosis causes cerebral infarction in the posterior circulation territory, as in this case. OBSERVATIONS: The patient's right PPHA had severe and progressive stenosis; therefore, he experienced cerebral infarction despite medical treatment. Therefore, PTA for the stenosis was performed, which ceased the recurrence of cerebral infarction and dizziness by improving blood flow in the posterior circulation. LESSONS: Several reports have described ICA stenosis accompanied by PPHA or PPHA stenosis in patients receiving endovascular treatments. Almost all cases were nonprogressive, and the treatment procedure was stenting. However, in our case, the PPHA stenosis was progressive, and we performed PTA because the patient experienced resistance to antiplatelet drugs and had poor collateral flow.
ABSTRACT
Background: The primitive hypoglossal artery (PHA) is an anastomotic vessel of the carotid-basilar artery system that is prevalent only transiently during the embryonic period. Persistent primitive hypoglossal artery (PPHA) is a rare vessel variation in which PHA exists persistently in adulthood and occurs in approximately 0.02-0.1% of the population. Tortuosity of the extracranial internal carotid artery (ICA) is relatively common, impacting 10-43% of the population, and is caused by either congenital or acquired factors. It is still unknown whether PPHA and tortuosity of extracranial ICA are associated. Here, we present a case report of the concurrence of three types of pathologies of the carotid artery: extreme coiling of the extracranial internal carotid artery, multiple aneurysms and persistent primitive hypoglossal artery. Case description: A 66-year-old woman suffered intermittent headaches, dizziness and numbness of the right eyelid for 5 years. Magnetic resonance angiography performed in a local hospital reported an aneurysm of the posterior communicating artery segment of the left ICA and a left PPHA. Digital subtraction angiography conducted after admission showed a PPHA originating from the left cervical ICA and an extremely coiling segment of the ICA distal to the beginning of PPHA. Except for the aneurysm of the posterior communicating artery segment of the left ICA, multiple aneurysms were found at the coiling segment of the ICA. Conclusion: To the best of our knowledge, this is the first report of PPHA accompanied by an adjacent, extremely coiling ICA. There are no reports of similar tortuous ICAs to this extent or at this position. Including aneurysms, three types of pathologies suggest their congenital origin, and a review of the literature infers the probable association of these lesions.
ABSTRACT
Persistent primitive hypoglossal artery (PHA) originating from the external carotid artery (ECA) is a rare anomaly. Reports of carotid endarterectomy (CEA) for ECA stenosis associated with this anomaly are even rarer. A 76-year-old woman presented to a medical clinic with a major complaint of refractory dizziness. Carotid ultrasound study suggested severe stenosis of the left cervical carotid bifurcation; therefore, she was referred to our department for a possible CEA. The imaging results indicated severe stenosis of the left carotid bifurcation and that the ECA was a PHA and the origin of the dominant vertebrobasilar artery (VBA). CEA was performed with the special caution of providing VBA collateral flow during clamping and preventing microembolisms during declamping of the ECA. Postoperative head magnetic resonance imaging revealed no new findings of cerebral infarction, and her dizziness disappeared. CEA associated with stenosis of the PHA as the origin of a dominant VBA was safely performed with an appropriate understanding of possible collateral pathways during cross-clamping.
Subject(s)
Carotid Stenosis , Endarterectomy, Carotid , Vertebrobasilar Insufficiency , Aged , Carotid Artery, Internal/diagnostic imaging , Carotid Artery, Internal/surgery , Carotid Stenosis/complications , Carotid Stenosis/diagnostic imaging , Carotid Stenosis/surgery , Constriction, Pathologic , Dizziness/complications , Endarterectomy, Carotid/adverse effects , Female , Humans , Vertebrobasilar Insufficiency/complications , Vertebrobasilar Insufficiency/diagnostic imagingABSTRACT
Only several cases of internal carotid artery (ICA) stenosis involving the persistent primitive hypoglossal artery (PPHA) have been treated with carotid endarterectomy (CEA) because of its extreme rarity. CEA was performed for an 87-year-old female with severe stenosis of the right ICA-PPHA bifurcation requiring shunting from CCA to both PPHA and ICA. We initially attempted to insert two intraluminal balloon shunts into the CCA, as previously reported. However, we found this procedure technically impossible to achieve. An improvised three-way junction tube was inserted distally into PPHA and ICA and proximally into CCA, securing blood flow during CEA. Unfortunately, the patient suffered post-operative ischemic brain lesions due to the prolonged ischemic time during our initial unsuccessful shunt attempt. A three-way junction shunting tube could be an effective shunt technique during an anatomically complicated CEA.
ABSTRACT
OBJECTIVE: Persistent primitive hypoglossal artery (PPHA) is a rare type of persistent carotid-basilar anastomosis sometimes associated with other vascular lesions. We treated an extremely rare case of PPHA with concomitant ipsilateral symptomatic cervical internal carotid artery (ICA) stenosis and unruptured aneurysm. CASE PRESENTATION: A 67-year-old woman visited our institution with acute onset of diplopia. Magnetic resonance imaging revealed multiple acute infarctions in the right anterior and posterior circulations. Digital subtraction angiography demonstrated the right PPHA concomitant with ipsilateral cervical ICA stenosis and an unruptured ICA aneurysm with maximum diameter of 8 mm. The multiple infarctions were considered to result from artery-to-artery embolism due to microthrombi from the ICA plaque passed along the PPHA, so carotid endarterectomy was performed as the first step with preoperative modified Rankin Scale (mRS) grade 1. During the operation, the patient had impaired ICA perfusion due to internal shunt catheter migration into the PPHA followed by acute infarction in the right hemisphere causing mild left hemiparesis. The patient was transferred to the rehabilitation hospital with mRS grade 3. After 3 months of rehabilitation, the patient recovered to mRS grade 1 and clipping surgery for the unruptured right ICA aneurysm was performed as the second step with uneventful postoperative course. CONCLUSION: The treatment strategy should be carefully considered depending on the specific blood circulation for such cases of PPHA with unique vasculature.
Subject(s)
Basilar Artery , Carotid Arteries , Carotid Stenosis , Intracranial Aneurysm , Aged , Angiography , Basilar Artery/abnormalities , Basilar Artery/diagnostic imaging , Carotid Arteries/abnormalities , Carotid Arteries/diagnostic imaging , Carotid Stenosis/diagnostic imaging , Female , Humans , Intracranial Aneurysm/diagnostic imaging , Magnetic Resonance ImagingABSTRACT
Persistent primitive hypoglossal artery is a rare variant of intrauterine anastomosis between the carotid and basilar arteries, which may remain in adults. The presence of this artery in carotid artery atherosclerosis increases the risks for stroke in the carotid and basilar basins. Our clinical case illustrates successful carotid endarterectomy in the presence of an ipsilateral persistent primitive hypoglossal artery under cerebral oximetry control.
Subject(s)
Carotid Stenosis , Cerebrovascular Circulation , Adult , Carotid Artery, Internal , Carotid Stenosis/complications , Carotid Stenosis/diagnosis , Carotid Stenosis/surgery , Constriction, Pathologic , Humans , OximetryABSTRACT
Successful embolization of a basilar bifurcation aneurysm associated with a persistent primitive hypoglossal artery (PPHA) using Y-stent-assisted coiling.
ABSTRACT
BACKGROUND: Ruptured aneurysms associated with a partial vertebrobasilar duplication or a persistent primitive hypoglossal artery (PPHA) have been reported. Only rarely has endovascular treatment of ruptured aneurysms in association with both vascular variations been reported. OBSERVATIONS: A 66-year-old woman experienced the sudden onset of a severe headache caused by a subarachnoid hemorrhage. Cerebral angiograms demonstrated a prominent PPHA originating from the left internal carotid artery at the C2 vertebral level and a partial vertebrobasilar duplication between the hypoplastic right vertebral artery and proximal basilar artery with a small aneurysm at the proximal end of the duplication from where the anterior spinal artery originated. The left vertebral artery was aplastic. A microcatheter was introduced into the aneurysm via the PPHA under the control of high blood flow, using a balloon-assisted technique. The aneurysm was completely obliterated with a coil. Although small cerebellar and cerebral infarcts developed during the procedure, the patient was discharged without neurological symptoms. LESSONS: To avoid serious neurological complications, precise analysis of the complex vascular anatomy, including the anterior spinal artery and hemodynamics, is clinically important for endovascular therapy of cerebral aneurysms in patients with an association between a partial vertebrobasilar duplication and a PPHA.
ABSTRACT
Persistent primitive hypoglossal artery (PPHA) is a rare form of persistent embryonic carotid-basilar anastomosis. We present an unusual case of PPHA and an anterior choroidal artery (AChoA) aneurysm associated with Chiari type I malformation. A 45-year-old woman presented with transient dizziness. Magnetic resonance imaging revealed Chiari type I malformation and a left AChoA aneurysm. Digital subtraction angiography incidentally revealed a left PPHA. To the best of our knowledge, this is the first reported case of Chiari malformation in conjunction with PPHA and aneurysms. In this case, the perfusion of the posterior circulation is completely dependent on PPHA. It is very important to identify such variant vessels and complex angioarchitecture before planning neuroendovascular or surgical intervention to prevent possible risks.
ABSTRACT
A 51-year-old Japanese woman was admitted to our hospital because of speech difficulty following severe headache. Neurological examination showed dysarthria and tongue weakness on the right side, indicating right hypoglossal nerve palsy. Needle electromyography of the right side of the tongue showed fibrillation potentials. Magnetic resonance angiography and computed tomography angiography revealed a right, persistent, primitive hypoglossal artery (PPHA) that met Lie's diagnostic criteria. Digital subtraction angiography showed an extended PPHA with irregular caliber in the portion running through the right hypoglossal canal. We diagnosed compression neuropathy of the hypoglossal nerve due to PPHA enlargement based on the findings of ipsilateral hypoglossal nerve palsy, fibrillation that indicated peripheral nerve palsy, and the enlarged diameter of the portion of the PPHA running through the right hypoglossal canal. We prescribed antihypertensive therapy. At 1 year after onset, her tongue weakness was alleviated. Clinicians should consider compression neuropathy due to a PPHA as one of the possibilities in the differential diagnosis of hypoglossal nerve palsy.
Subject(s)
Arteries/abnormalities , Hypoglossal Nerve Diseases/etiology , Hypoglossal Nerve/physiopathology , Nerve Compression Syndromes/etiology , Tongue/blood supply , Tongue/innervation , Vascular Malformations/complications , Antihypertensive Agents/therapeutic use , Arteries/diagnostic imaging , Arteries/physiopathology , Female , Humans , Hypoglossal Nerve Diseases/diagnosis , Hypoglossal Nerve Diseases/physiopathology , Middle Aged , Nerve Compression Syndromes/diagnosis , Nerve Compression Syndromes/physiopathology , Recovery of Function , Treatment Outcome , Vascular Malformations/diagnostic imaging , Vascular Malformations/drug therapy , Vascular Malformations/physiopathologyABSTRACT
Carotid mycotic aneurysm is extremely rare and even more unusual when it is associated with a persistent primitive hypoglossal artery. This artery is the second most common of the embryonic carotid-vertebrobasilar anastomoses. It originates from the cervical internal carotid artery and enters the cranium through a widened hypoglossal canal before anastomosing with the basilar artery. We report a case of an elderly man with a rare Salmonella-induced mycotic aneurysm associated with a persistent primitive hypoglossal artery. Surgical resection of the mycotic aneurysm was complicated by a posterior circulation stroke. To the best of our knowledge, there was no previous report of a carotid mycotic aneurysm associated with a persistent primitive hypoglossal artery thus far in the literature. Owing to the high mortality rate of the carotid mycotic aneurysm, it is imperative to be familiar with the vascular and imaging anatomy prior to surgery particularly in the presence of an embryonic carotid-vertebrobasilar anastomosis. In this report, we highlight the imaging characteristics and treatment options for this rare mycotic aneurysm together with a literature review.
Subject(s)
Aneurysm, Infected/diagnostic imaging , Aneurysm, Infected/pathology , Basilar Artery/abnormalities , Carotid Artery, Internal/abnormalities , Salmonella Infections/diagnostic imaging , Salmonella Infections/pathology , Aged , Aneurysm, Infected/surgery , Angiography , Basilar Artery/diagnostic imaging , Carotid Artery, Internal/diagnostic imaging , Diagnosis, Differential , Humans , Male , Postoperative Complications , Salmonella Infections/surgery , Stroke/etiology , Tomography, X-Ray ComputedABSTRACT
We report a case of an anomalous anastomosis formed between the external carotid artery (ECA) and the vertebrobasilar artery (VBA) and passing through the hypoglossal canal. A carotid-vertebrobasilar anastomosis of this kind is typically considered a variant of persistent primitive hypoglossal artery which usually originates from the internal carotid artery. However, the anastomotic vessel in this case had a common trunk with the occipital artery (OA), a remnant of the primitive proatlantal artery. The proximal and distal parts of the anastomotic vessel seemed to have been derived from the primitive proatlantal artery and the primitive hypoglossal artery, respectively. Thus, we propose that this ECA-VBA anastomosis, which passed through the hypoglossal canal and had a common trunk with the OA, be referred to as a dilated primitive hypoglossal-proatlantal anastomosis; that is, a dilated ascending pharyngeal artery rather than a variant of persistent primitive hypoglossal artery.
Subject(s)
Arterio-Arterial Fistula/diagnostic imaging , Carotid Artery, External/abnormalities , Occipital Bone/blood supply , Vertebral Artery/abnormalities , Arterio-Arterial Fistula/etiology , Carotid Artery, External/diagnostic imaging , Female , Humans , Magnetic Resonance Angiography , Middle Aged , Occipital Bone/diagnostic imaging , Vertebral Artery/diagnostic imagingABSTRACT
Persistent primitive hypoglossal artery is a rare anastomosis between the carotid and basilar arteries, and sometimes associated with cerebral aneurysms. However, association of persistent primitive hypoglossal artery with aneurysms located on arteries other than persistent primitive hypoglossal artery itself or posterior circulation is very rare. An 80-year-old woman suffered from subarachnoid hemorrhage, whose angiography demonstrated aneurysms on the left middle cerebral artery and anterior communicating artery, and the left persistent primitive hypoglossal artery. The middle cerebral artery aneurysm was the origin of hemorrhage. Although repeated craniotomy was necessary for the left middle cerebral artery aneurysm, both aneurysms were successfully clipped. In our case, neither aneurysm was located on an artery related to the persistent primitive hypoglossal artery. There is a possibility that cases of persistent primitive hypoglossal artery are accompanied by cerebral aneurysms on arteries other than the persistent primitive hypoglossal artery or in the posterior circulation.
ABSTRACT
Persistent primitive hypoglossal artery is a carotid-vertebrobasilar anastomosis, which commonly arises from the internal carotid artery at the level of the C (cervical) 1-3 vertebrae. We describe a unique case of a female infant patient with this anomaly that has an unusually low origin from the distal common carotid artery just below the bifurcation at the level of roughly C5 and supplies the entire vertebrobasilar system. Additional cardiovascular anatomical variations were present: Tetralogy of Fallot and a right-sided aortic arch with mirror image branching. These singular variations are rare in the general population, but even rarer when combined. Awareness of these unusual vascular variants is clinically significant, as they may predispose the patients to early ischemic injury, hemorrhage, aneurysm formation, and can be essential in surgical planning. Therefore, radiographic imaging is of importance in proper diagnosis of such variants.
Subject(s)
Abnormalities, Multiple/diagnostic imaging , Basilar Artery/abnormalities , Basilar Artery/diagnostic imaging , Carotid Artery, Internal/abnormalities , Carotid Artery, Internal/diagnostic imaging , Vertebral Artery/abnormalities , Vertebral Artery/diagnostic imaging , Aorta, Thoracic/abnormalities , Aorta, Thoracic/diagnostic imaging , Female , Humans , Infant, Newborn , Magnetic Resonance Angiography , Tetralogy of Fallot/diagnostic imagingABSTRACT
BACKGROUND: Persistent carotid-basilar connections have a prevalence of 0.14%. Recognizing such persistent fetal anastomoses between the carotid and the vertebrobasilar circulation is of great importance because they are reportedly associated with an increased prevalence of intracranial aneurysms. METHODS: We report the case of a 15-year-old female patient who presented with a World Federation of Neurosurgical Societies grade 5 subarachnoid hemorrhage from an aneurysm at the junction of a persistent primitive hypoglossal artery and the posterior inferior cerebellar artery origin. Supratentorially, unfortunately, there was no parenchymal blush or cortical venous return. Eventually, a multidisciplinary decision was made to withdraw care. RESULTS: Fifty-seven cases were reported in the literature to date of persistent hypoglossal arteries, 16 of which presented with an associated aneurysm, 5 with an arteriovenous malformation, and 6 with a subarachnoid hemorrhage. Our case is the youngest patient reported so far. Hypoplasia or aplasia of the vertebral artery often were encountered (36 and 13 cases, respectively), as well as carotid artery stenosis (15 cases). CONCLUSIONS: Although uncommon, it is important to recognize persistent carotid-basilar connections, since they have a considerable hemodynamic impact on the posterior cerebral circulation via the carotid system. A critical reduction in the carotid blood flow will, therefore, have ischemic consequences in the posterior cerebral territories. In addition, such connections might be associated with anomalies of the vessel wall and be predisposed to aneurysm formation. The endovascular neurointerventionalist, as well as the vascular and skull base neurosurgeon, need to be aware of their anatomy and variations.