Percutaneous transluminal angioplasty for persistent primitive hypoglossal artery stenosis: illustrative case

Katsuma Iwaki Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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Koichi Arimura Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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Shunichi Fukuda Department of Neurosurgery, National Hospital Organization Kyushu Medical Center, Fukuoka, Japan; and

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Soh Takagishi Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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Ryota Kurogi Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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Kuniyuki Nakamura Department of Medicine and Clinical Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan

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Akira Nakamizo Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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Koji Yoshimoto Department of Neurosurgery, Kyushu University, Fukuoka, Japan

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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.

ABBREVIATIONS

BA = basilar artery; CEA = carotid endarterectomy; ECA = external carotid artery; ICA = internal carotid artery; LDL-c = low-density lipoprotein cholesterol; MRI = magnetic resonance imaging; PCoA = posterior communicating artery; PPHA = persistent primitive hypoglossal artery; PRU = P2Y12 reaction unit; PTA = percutaneous transluminal angioplasty; rSO2 = regional cerebral oxygenation; VA = vertebral artery

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.

ABBREVIATIONS

BA = basilar artery; CEA = carotid endarterectomy; ECA = external carotid artery; ICA = internal carotid artery; LDL-c = low-density lipoprotein cholesterol; MRI = magnetic resonance imaging; PCoA = posterior communicating artery; PPHA = persistent primitive hypoglossal artery; PRU = P2Y12 reaction unit; PTA = percutaneous transluminal angioplasty; rSO2 = regional cerebral oxygenation; VA = vertebral artery

A persistent primitive hypoglossal artery (PPHA) is a type of carotid-basilar anastomosis with an incidence of 0.02% to 0.10%.1 It arises from the extracranial internal carotid artery (ICA) at the C1 to C3 level and connects to the basilar artery (BA) via the hypoglossal canal. It can be associated with hypoplasia or the absence of the ipsilateral posterior communicating artery (PCoA) and vertebral artery (VA); therefore, its stenosis or occlusion causes serious cerebral infarction in the posterior circulation area.2 Several cases of ICA stenosis accompanied by PPHA or PPHA stenosis have been treated surgically and endovascularly; however, we did not find reports regarding endovascular treatment for progressive stenosis of the PPHA.3,4 Here, we describe for the first time a case of symptomatic and progressive stenosis of a PPHA that was treated using percutaneous transluminal angioplasty (PTA).

Illustrative Case

History and Examination

A 68-year-old male with trigeminal neuralgia was referred to our hospital. The patient and his family had no history of cerebrovascular diseases, and he was not receiving any medication. Cranial and cervical magnetic resonance imaging (MRI) showed acute cerebral infarction in the right occipital lobe and stenosis of the right ICA and PPHA (Fig. 1A). His serum low-density lipoprotein cholesterol (LDL-c) level was 204 mg/dL, and he had never received medical treatment; therefore, we first administered aspirin and statins. His serum LDL-c level decreased to 70 mg/dL; however, he had a recurrent cerebral infarction in his left occipital lobe 5 months after the first treatment (Fig. 1B); therefore, prasugrel was added. Despite the additional treatment, the patient developed another cerebral infarction in his right cerebellum (Fig. 1C). Moreover, he presented with dizziness, and MRI revealed progressive stenosis of the PPHA. Collateral flow was not observed when his PPHA was occluded because his bilateral PCoA was hypoplastic, the left subclavian artery was constricted, so the blood flow of the posterior circulation seemed to be insufficient only by the left VA (Fig. 2). Therefore, PTA and stenting of the PPHA were planned.

FIG. 1
FIG. 1

Axial diffusion-weighted image of cranial MRI and cervical magnetic resonance angiography (MRA). A: MRI and MRA at the first visit to our hospital. The patient had an acute cerebral infarction in the right occipital lobe, stenosis of the right ICA, and PPHA. B: MRI and MRA at 5 months after the first visit. There was recurrent cerebral infarction in the left occipital lobe. The PPHA had worsened (arrows). C: MRI and MRA at 7 months after the first visit. There was recurrent cerebral infarction in the right cerebellum. The PPHA had nearly disappeared (arrows).

FIG. 2
FIG. 2

A: Image from the three-dimensional computed tomography angiography (3D-CTA), as seen from above. The bilateral PCoAs were not visable. B: Anteroposterior view of the left subclavian artery (SCA) angiogram. There was stenosis of the left SCA, and the left VA was hypoplastic. C: Anteroposterior intracranial view of the left SCA angiogram. Ant = anterior; Post = posterior.

Treatment

First, we evaluated platelet reactivity using VerifyNow Aspirin and P2Y12 assays (Instrumentation Laboratory). The level of his aspirin reaction units was 402, his P2Y12 reaction unit (PRU) was 237, and the percentage of platelet inhibition was 8%. Therefore, the efficacy of prasugrel was insufficient. Hence, we added cilostazol (200 mg) before surgery.

The procedure was performed with the patient under local anesthesia, and regional cerebral oxygenation (rSO2) in the frontal lobes was monitored using near-infrared spectroscopy during the intervention. A 9-Fr long sheath was placed in the right common femoral artery. We planned to perform the flow-reversal technique using a double-balloon guiding system (Mo.Ma Ultra, Medtronic). The proximal balloon was placed at the ICA bifurcation, and the distal balloon was placed in the petrous portion of the ICA (Fig. 3B). When the proximal balloon was inflated, his right rSO2 decreased from 58% to 30%, and he lost consciousness; therefore, we discontinued the flow-reversal technique. We then placed distal filter protection (FilterWire EZ, Boston Scientific) in the distal PPHA (Fig. 3B). A 30-second angioplasty was performed using a balloon with a diameter of 3.5 mm (Fig. 3B). After dilatation, the blood flow of the PPHA markedly improved, and restenosis was not observed (Fig. 3C–E). Initially, we planned to stent the PPHA; however, since the stent jailed the ICA and the patient experienced resistance to platelet inhibitors of the P2Y12 receptor, we judged that there was a risk of ICA occlusion. Therefore, we decided not to perform stenting at that time.

FIG. 3
FIG. 3

Catheter angiogram during PTA for the PPHA. A: Anteroposterior view of the right common carotid artery (CCA) angiogram before angioplasty. There was severe stenosis at the origin of the PPHA, and the blood flow through the PPHA was quite slow. B: An instant image regarding the PTA procedure. The proximal balloon was placed at the ICA bifurcation, the distal balloon was placed at the distal ICA, and a filter wire was placed at the distal PPHA. Angioplasty was performed using a balloon with a diameter of 3.5 mm (arrows). C: Anteroposterior view of the right CCA angiogram after angioplasty. The stenosis and blood flow in the PPHA had improved. D: 3D digital subtraction angiography (3D-DSA) of the right CCA before angioplasty. E: 3D-DSA of the right CCA after angioplasty. Stenosis of the PPHA had improved (arrow). ECA = external carotid artery; Dist. = distal; Prox. = proximal.

The patient’s postoperative course was uneventful, with an improvement in dizziness. A 123I-N-isopropyl-iodoamphetamine single-photon emission computed tomography scan obtained on the day after surgery showed increased blood flow in the cerebellum and occipital lobe. The patient underwent follow-up MRI the following day and 7 days and 6 months later. No ischemic lesions or restenosis of the PPHA were observed (Fig. 4). He was experiencing no inconvenience in his daily life by his latest follow-up. In particular, the improved blood flow made it easy to manage his blood pressure.

FIG. 4
FIG. 4

Cervical MRA before and after angioplasty. A: MRA before angioplasty. B: MRA on the day after angioplasty. C: MRA at 7 days after angioplasty. D: MRA at 6 months after angioplasty. There was no restenosis.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

There are several types of carotid-vertebrobasilar anastomoses, including trigeminal, hypoglossal, otic, and proatlantal arteries.2 A PPHA arises from the cervical ICA and connects to the BA via the hypoglossal canal.1 Because the MRI of the patient showed that the vessel originating from the ICA passed through the hypoglossal canal and was anastomosed to the BA, we could distinguish the present case from those of other types of anastomosis.

In most cases, the PPHA is the major blood supply to the posterior circulation because the VA and PCoA are often aplastic or hypoplastic, as in our case.1,2 Therefore, PPHA stenosis or occlusion can cause cerebral infarction in the posterior circulation area. After the angioplasty, the dizziness and recurrent infarction stopped, suggesting that the patient’s symptoms were possibly flow related.

There are some previous reports regarding carotid endarterectomy (CEA) for cases of ICA stenosis with PPHA.3,5–13 However, CEA for PPHA may be difficult because, in most cases, the PPHA is located at a high position. Therefore, endovascular treatment is effective in such cases.

Table 1 presents the case list of endovascular treatment for ICA stenosis with PPHA and PPHA stenosis.4,14–21 In our review of previous reports, the treatment for seven cases involved carotid artery stenting for the ICA proximal to the origin of the PPHA. Only two cases were treated for PPHA stenosis.4,17 One involved stenting for the distal PPHA; therefore, the treatment modality was different from that in our case. The stenotic lesion reported by Li et al.4 was the origin of the PPHA, as in our case; however, our case was different in that the patient presented with some symptoms and experienced recurrent cerebral infarction and progressive stenosis of the PPHA. All cases in our review were treated with stenting; however, we avoided using stents because of antiplatelet drug resistance and the lack of collateral flow. As reported previously, the ICA is always jailed when using a stent for PPHA stenosis. Clinicians occasionally experience external carotid artery (ECA) occlusion after carotid stenting22 because the ECA is usually jailed by a stent. In most cases, ECA occlusion is usually not problematic. However, in our case, the jailed vessel was the ICA, and the occlusion caused a severe stroke. Thus, we believe that PTA was the best treatment method in our case.

TABLE 1

Literature review of endovascular treatment for ICA stenosis with PPHA and PPHA stenosis

Case No.Authors & YearAge (yrs), SexLocation of StenosisSymptomsInfarctionProgressionVAPCoATreatmentPicture FU
1Kanazawa et al., 20081468, MLt ICA stenosis (proximal to origin of PPHA)SyncopeNoneNoneBilat: absentNoneCASND
2Nii et al., 20101562, MRt ICA stenosis (proximal to origin of PPHA)Transient hand weaknessNoneNoneRt: absent, lt: hypoplasticNoneCAS3 mos, no restenosis
3Silva et al., 20131663, FRt ICA stenosis (proximal & at level of origin of PPHA)Transient hand weakness & numbnessNoneNoneNDNDCAS6 mos, no restenosis
4Eller et al., 201417Mid-60sRt ICA stenosis (proximal to origin of PPHA), rt PPHA stenosis (origin of PPHA)Transient face numbness, nausea & vomitingNoneNoneBilat: hypoplasticBilat: hypoplasticCAS for PPHA (distal of PPHA)ND
5Li et al., 2014447, MRt ICA & PPHA stenosisNoneNoneNoneBilat: hypoplasticBilat: hypoplasticStenting for PPHA (PPHA to ICA)Rt ICA & PPHA stenosis
6Bikei et al., 20161860, FLt ICA stenosis (proximal to the origin of PPHA)NoneNoneNoneRt: hypoplastic, lt: normalRt: none, lt: infundibularCASND
7Murai et al., 20161977, MRt ICA stenosis (proximal to origin of PPHA)NoneRt MCA territoryICA stenosisRt: absent, lt: hypoplasticNoneCASND
8Burgard et al. 20202078, MLt ICA stenosis (proximal to origin of PPHA)Rt hemiparesisLt MCA territoryNoneBilat: hypoplasticNDCASND
9Shehab et al. 20232177, FRt ICA stenosis (proximal to origin of PPHA)Weakness of both legs & armsPonsNoneNDNDCASND
10Present case68, MRt PPHA stenosis (origin of PPHA)DizzinessBilat occipital lobe, rt cerebellumPPHA stenosisRt: absent, lt: hypoplasticBilat: hypoplasticPTA for PPHA6 mos, no restenosis

CAS = carotid artery stenting; FU = follow-up; MCA = middle cerebral artery; ND = not described.

Lessons

To the best of our knowledge, this is the first reported case of progressive PPHA stenosis that caused recurrent cerebral infarction requiring PTA. If the PPHA originates from a stenotic lesion, stenting risks ICA occlusion due to stent jailing. Because the patient in our case was antiplatelet-resistant, the risk of occlusion was considered higher than usual. In such cases, PTA alone may be sufficient without stenting.

Acknowledgments

We thank the individuals who contributed to the study or manuscript preparation but did not fulfill all criteria for authorship.

Author Contributions

Conception and design: Iwaki, Arimura. Acquisition of data: Iwaki, Fukuda, Takagishi. Analysis and interpretation of data: Iwaki. Drafting the article: Iwaki. Critically revising the article: Iwaki, Nakamizo. Reviewed submitted version of manuscript: Iwaki, Kurogi, Nakamura. Approved the final version of the manuscript on behalf of all authors: Iwaki. Administrative/technical/material support: Iwaki. Study supervision: Arimura, Takagishi, Kurogi, Yoshimoto.

References

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    Sanada T, Shirai W, Yamamoto S, Kinoshita M, Tokumitsu N. A case of carotid endarterectomy assisted with a three-way junction shunting tube for the internal carotid artery stenosis involving a persistent primitive hypoglossal artery. J Surg Case Rep. 2021;2021(8):rjab362.

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    Eller JL, Jahshan S, Dumont TM, Kan P, Siddiqui AH. Tandem symptomatic internal carotid artery and persistent hypoglossal artery stenosis treated by endovascular stenting and flow reversal. BMJ Case Rep. 2013;2013:bcr2012010578.

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  • FIG. 1

    Axial diffusion-weighted image of cranial MRI and cervical magnetic resonance angiography (MRA). A: MRI and MRA at the first visit to our hospital. The patient had an acute cerebral infarction in the right occipital lobe, stenosis of the right ICA, and PPHA. B: MRI and MRA at 5 months after the first visit. There was recurrent cerebral infarction in the left occipital lobe. The PPHA had worsened (arrows). C: MRI and MRA at 7 months after the first visit. There was recurrent cerebral infarction in the right cerebellum. The PPHA had nearly disappeared (arrows).

  • FIG. 2

    A: Image from the three-dimensional computed tomography angiography (3D-CTA), as seen from above. The bilateral PCoAs were not visable. B: Anteroposterior view of the left subclavian artery (SCA) angiogram. There was stenosis of the left SCA, and the left VA was hypoplastic. C: Anteroposterior intracranial view of the left SCA angiogram. Ant = anterior; Post = posterior.

  • FIG. 3

    Catheter angiogram during PTA for the PPHA. A: Anteroposterior view of the right common carotid artery (CCA) angiogram before angioplasty. There was severe stenosis at the origin of the PPHA, and the blood flow through the PPHA was quite slow. B: An instant image regarding the PTA procedure. The proximal balloon was placed at the ICA bifurcation, the distal balloon was placed at the distal ICA, and a filter wire was placed at the distal PPHA. Angioplasty was performed using a balloon with a diameter of 3.5 mm (arrows). C: Anteroposterior view of the right CCA angiogram after angioplasty. The stenosis and blood flow in the PPHA had improved. D: 3D digital subtraction angiography (3D-DSA) of the right CCA before angioplasty. E: 3D-DSA of the right CCA after angioplasty. Stenosis of the PPHA had improved (arrow). ECA = external carotid artery; Dist. = distal; Prox. = proximal.

  • FIG. 4

    Cervical MRA before and after angioplasty. A: MRA before angioplasty. B: MRA on the day after angioplasty. C: MRA at 7 days after angioplasty. D: MRA at 6 months after angioplasty. There was no restenosis.

  • 1

    Oelerich M, Schuierer G. Primitive hypoglossal artery: demonstration with digital subtraction-, MR- and CT angiography. Eur Radiol. 1997;7(9):14921494.

  • 2

    Menshawi K, Mohr JP, Gutierrez J. A functional perspective on the embryology and anatomy of the cerebral blood supply. J Stroke. 2015;17(2):144158.

  • 3

    Thayer WP, Gaughen JR, Harthun NL. Surgical revascularization in the presence of a preserved primitive carotid-basilar communication. J Vasc Surg. 2005;41(6):10661069.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Zhang L, Song G, Chen L, Jiao L, Chen Y, Wang Y. Concomitant asymptomatic internal carotid artery and persistent primitive hypoglossal artery stenosis treated by endovascular stenting with proximal embolic protection. J Vasc Surg. 2016;63(1):237240.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Sunada I, Yamamoto S, Matsuoka Y, Nishimura S. Endarterectomy for persistent primitive hypoglossal artery--case report. Neurol Med Chir (Tokyo). 1991;31(2):104108.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Megyesi JF, Findlay JM, Sherlock RA. Carotid endarterectomy in the presence of a persistent hypoglossal artery: case report. Neurosurgery. 1997;41(3):669672.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Hatayama T, Yamane K, Shima T, Okada Y, Nishida M. Persistent primitive hypoglossal artery associated with cerebral aneurysm and cervical internal carotid artery stenosis--case report. Neurol Med Chir (Tokyo). 1999;39(5):372375.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Kawabori M, Kuroda S, Yasuda H, et al. Carotid endarterectomy for internal carotid artery stenosis associated with persistent primitive hypoglossal artery: efficacy of intraoperative multi-modality monitoring. Minim Invasive Neurosurg. 2009;52(5-6):263266.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Pride LB, Lagergren ER, Hafner DH, Chervu AA. Critical carotid artery stenosis involving a persistent primitive hypoglossal artery. J Vasc Surg Cases Innov Tech. 2020;6(2):177180.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Kawamura K, Tokugawa J, Watanabe M, et al. Persistent primitive hypoglossal artery with ipsilateral symptomatic carotid artery stenosis and cerebral aneurysm. J Stroke Cerebrovasc Dis. 2021;30(11):106099.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Sanada T, Shirai W, Yamamoto S, Kinoshita M, Tokumitsu N. A case of carotid endarterectomy assisted with a three-way junction shunting tube for the internal carotid artery stenosis involving a persistent primitive hypoglossal artery. J Surg Case Rep. 2021;2021(8):rjab362.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Segawa M, Inoue T, Tsunoda S, Noda R, Akabane A. Carotid endarterectomy for vertebrobasilar insufficiency caused by severe stenosis of primitive hypoglossal artery: a technical case report and literature review. Neurol Med Chir (Tokyo). 2022;62(5):254259.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Telianidis S, Westcott MJ, Ironfield CM, Sanders LM. Case of amaurosis fugax in the setting of a persistent primitive hypoglossal artery requiring carotid endarterectomy with regional anesthesia. Am J Case Rep. 2023;24:e939450.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Kanazawa R, Ishihara S, Okawara M, Ishihara H, Kohyama S, Yamane F. A successful treatment with carotid arterial stenting for symptomatic internal carotid artery severe stenosis with ipsilateral persistent primitive hypoglossal artery: case report and review of the literature. Minim Invasive Neurosurg. 2008;51(5):298302.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

    Nii K, Aikawa H, Tsutsumi M, et al. Carotid artery stenting in a patient with internal carotid artery stenosis and ipsilateral persistent primitive hypoglossal artery presenting with transient ischemia of the vertebrobasilar system: case report. Neurol Med Chir (Tokyo). 2010;50(10):921924.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Silva CF, Hou SY, Kühn AL, Whitten RH, Wakhloo AK. Double embolic protection during carotid artery stenting with persistent hypoglossal artery. BMJ Case Rep. 2013;2013:bcr2013010709.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Eller JL, Jahshan S, Dumont TM, Kan P, Siddiqui AH. Tandem symptomatic internal carotid artery and persistent hypoglossal artery stenosis treated by endovascular stenting and flow reversal. BMJ Case Rep. 2013;2013:bcr2012010578.

    • PubMed
    • Search Google Scholar
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  • 18

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