- Email: [email protected]
Surgical Revascularization: Ligation of Extracranial Internal Carotid Artery and Superficial Temporal Artery-to-Middle Cerebral Artery Bypass in Patient with Extracranial Internal Carotid Aneurysm and Hemorrhagic Moyamoya Disease
Case Report
Surgical Revascularization: Ligation of Extracranial Internal Carotid Artery and Superficial Temporal Artery-to-Middle Cerebral Artery Bypass in Patient with Extracranial Internal Carotid Aneurysm and Hemorrhagic Moyamoya Disease Qingdong Han, Peng Zhou, Yabo Huang
Key words Extracranial internal carotid artery aneurysm - Moyamoya disease - Surgical revascularization -
Abbreviations and Acronyms CT: Computed tomography CTA: Computed tomography angiography CTP: Computed tomography perfusion DSA: Digital subtraction angiography EICA: Extracranial internal carotid artery HMMD: Hemorrhagic moyamoya disease ICA: Internal carotid artery MMD: Moyamoya disease MRI: Magnetic resonance imaging STA-MCA: Superficial temporal artery-to-middle cerebral artery Department of Neurosurgery, the First Affiliated Hospital of Soochow University, Suzhou, Jiangsu, China To whom correspondence should be addressed: Yabo Huang, M.D. [E-mail: [email protected]] Qingdong Han and Peng Zhou contributed equally to this article are considered as coefirst authors. Citation: World Neurosurg. (2019) 126:129-133. https://doi.org/10.1016/j.wneu.2019.02.110
- BACKGROUND:
Extracranial internal carotid artery (EICA) aneurysm is regarded as a rare lesion. What is more, patients can suffer from EICA aneurysms and hemorrhagic moyamoya disease (MMD) simultaneously.
- CASE
DESCRIPTION: A 38-year-old man was admitted to our hospital and underwent emergent tracheal intubation for hoarseness and breathing difficulty for 1 day. He had a pulsating mass of 2.5 cm in diameter in the left cervical area. Imaging examinations revealed the left EICA aneurysm and MMD. The patient underwent left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass and ligation of the left EICA simultaneously. Computed tomography revealed hemorrhagic stroke in the right basal ganglia region 20 days after the bypass. The hematoma disappeared 20 days after the stroke. Right bypass was performed 6 months after the left bypass. The patient was discharged with no neurologic abnormalities 10 days after the right bypass. He lived a normal life, and no neurologic abnormalities were found at the 6-month follow-up. Imaging examinations indicated the efficacy of the bilateral STA-MCA bypass and verified the disappearance of the left EICA aneurysm after the ligation.
- CONCLUSIONS:
We present a case of EICA aneurysm and hemorrhagic MMD. Ligation of the left ECA eliminates the EICA aneurysm with no neurologic abnormalities. STA-MCA bypass can be regarded as an effective choice for hemorrhagic MMD. Further studies should be performed to confirm the effectiveness of combined surgical revascularization.
Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION Extracranial internal carotid artery (EICA) aneurysm is regarded as a rare lesion. EICA aneurysms account for less than 1% of all peripheral artery aneurysms.1 EICA aneurysm may lead to ischemic stroke, cranial nerve compression, and other severe effects.2 Limited guidelines can be used for the treatment of EICA aneurysms. What is more, if patients suffer EICA aneurysm and hemorrhagic moyamoya disease (HMMD) simultaneously, the complicated neck mass and intracranial lesions can make neurosurgeons feel overwhelmed and helpless because recent treatments concerning HMMD are lacking and, when existing, controversial.
We present a patient with EICA aneurysm and HMMD simultaneously who was treated with surgical revascularization of ligation of the EICA and superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass. To the best of our knowledge, the present study is the first report regarding EICA aneurysm and HMMD simultaneously treated with surgical revascularization of ligation of EICA and STA-MCA bypass. CASE DESCRIPTION The study received consent from the ethics committee of the First Affiliated of Soochow University. It also was approved by the patient and his family. A 38-year-old man was admitted to emergency department of our hospital and underwent emergent tracheal intubation for hoarseness and
WORLD NEUROSURGERY 126: 129-133, JUNE 2019
breathing difficulty for 1 day. Before the onset of his hoarseness, the patient had intermittent headache for more than 3 years. He had no history of diabetes, hypertension, cerebral stroke, head and neck injuries or tumors, alcohol abuse, and smoking. Findings of the physical examination indicated that he had a left cervical pulsating mass of 2.5 cm in diameter. Computed tomography (CT), computed tomography angiography (CTA), digital subtraction angiography (DSA), magnetic resonance imaging (MRI), and computed tomography perfusion (CTP) imaging of the head and neck revealed the EICA aneurysm (Figure 1AeG) and MMD (Figure 1GeK). CT, CTA, and DSA indicated the left EICA aneurysm (Figure 1AeG). DSA showed moyamoya disease (MMD) in the patient (Figure 1GeH). In addition, DSA showed
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REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 1. Computed tomography (CT), computed tomography angiography (CTA), digital subtraction angiography (DSA), magnetic resonance imaging (MRI), and computed tomography perfusion (CTP) imaging of extracranial internal carotid artery aneurysm (AeG) and hemorrhagic moyamoya disease (GeK). (AeG) CT, CTA, and DSA indicating left extracranial internal
the anastomoses of the posterior communicating artery (Figure 1I) between the frontal segments of the Willis circle and the latter segments of the Willis circle in the patient with MMD. No masses or ischemic lesions were found on CTP (Figure 1J) or MRI (Figure 1K). Two days after admission, the patient underwent left STA-MCA bypass and the ligation of the left EICA simultaneously in our Neurosurgery Department. Postoperative CTA and DSA indicated the validity of the left STA-MCA bypass in our patient (Figure 2AeC) and the disappearance of the left EICA aneurysm after the ligation (Figure 2A) 4 days following the operations, simultaneously (Figure 2AeC). No ischemic lesions were found on the postoperative CTP (Figure 2C) following the aforementioned operations. However, the patient suffered deteriorated muscle strength from 5 to 2 grades in the left limbs 20 days following the aforementioned operations. Emergency CT showed hemorrhagic stroke in the right basal ganglia region 10 days after the left STA-MCA bypass (Figure 3A). After medical treatment of 20 days, the hematoma disappeared 20 days after the onset of the
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carotid artery aneurysm. (GeH) DSA showing moyamoya disease in the patient. What’s more, the arrow in the DSA shows the anastomoses (I) between the front segment of the Willis circle and the latter segment of the Willis circle in the patient with moyamoya disease. No ischemic lesions could be found on CTP (J) and MRI (K).
hemorrhagic stroke (Figure 3B) and the muscle strength in the left limbs returned to 4 grades. Right STA-MCA bypass was carried out 6 months after the left STA-MCA bypass (Figure 3C). The patient was discharged with no neurologic abnormalities 10 days after the right STAMCA bypass. No antiplatelet or anticoagulant measurements were performed after the patient was discharged from hospital. Follow-up The patient lived a normal life and no neurologic abnormalities were found at the 6-month follow-up after being discharged. DSA and CTP indicated the efficacy of the bilateral STA-MCA bypass (Figure 3DeF) and verified the disappearance of the left ECIA aneurysm after the ligation. DISCUSSION The case of ECIA aneurysm and HMMD is complicated and rare. The percentage of ECIA aneurysms to all peripheral artery aneurysms is less than 1%.1 The majority of patients with ECIA aneurysms have no symptoms, but the rest may suffer ischemic stroke, cranial nerve
compression, pulsatile cervical mass leading to dyspnea or dysphagia, pain, or even fatal hematemesis or epistaxis, of which transient ischemic attack is well accepted as the most common symptom.3,4 However, the high rate of mortality and stroke associated with ECIA aneurysms usually is found in earlier research.2 In the present study, our patient suffered no transient ischemic attack but had cranial nerve compression, and an emergent tracheal intubation for hoarseness and breathing difficulty was performed. The mass effect on the cranial nerve could be the chief culprit. The etiology of ECIA aneurysm is diverse. Atherosclerosis, iatrogenic or accidental injuries, fibromuscular dysplasia, EhlerseDanlos syndrome, and Marfan syndrome could be responsible for the occurrence of ECIA aneurysm.4,5 Usually, ECIA aneurysms can be classified into 2 subgroups, true ECIAs and pseudoaneurysms: the former account for 37%e 42% of ECIA aneurysms for atherosclerosis, and the latter aneurysms are caused by trauma, radiotherapy in the treatment of neck cancer, and carotid endarectomy.1 Atherosclerotic ECIA aneurysms usually are located in the proximal internal
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.110
CASE REPORT QINGDONG HAN ET AL.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 2. Computed tomography angiography (CTA) and digital subtraction angiography (DSA) indicating the validity of the left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass in a patient with moyamoya disease and the disappearance of the left extracranial internal carotid aneurysm after the ligation simultaneously (AeC). The thick arrows on CTA (A) and DSA (B) show the bypass. The thin arrow (A) indicates the disappearance of the left extracranial internal carotid aneurysm after the ligation. No ischemic lesions could be found on computed tomography perfusion (C) following the left STA-MCA bypass and ligation of the extracranial internal carotid artery.
carotid artery (ICA) and its bifurcation, but dysplastic aneurysms are located in the distal segment of the ICA and related to chronic dissection.3 In the present study, the ECIA aneurysm was located in the proximal segment of the ICA. Before the onset of his hoarseness, the patient had intermittent headache for more than 3 years. He had no history of diabetes, hypertension, cerebral stroke, head and neck injuries, tumors, alcohol abuse, and smoking. Moreover, as to the intermittent headache, no specific reasons could be found. As a relatively young patient with EICA aneurysm, no various inflammatory diseases were found. Which measures we can take toward EICA aneurysms may be still be limited and disputable. Some researchers advise that whatever it takes, surgical measures should be taken.6 Open surgery, endovascular
treatment, or medical treatment for the lesions can be taken into consideration. A previous study showed that direct ligation of the EICA of patients suffering EICA aneurysms accounted for 20%e40% postoperative severe central nervous system stroke or high mortality.7 In other words, if the surgeons attach little importance to the collateral circulation of the Willis circle, direct ligation of the ICA may be lifethreatening. Even some authors performed high-flow extracranialeintracranial bypass to treat EICA aneurysm.6 Regarding preoperative risk evaluation, the cerebral blood perfusion by balloon occlusion test with DSA was carried out, and no abnormalities occurred in the patient. With good collateral circulation posterior communicating artery between the front segment and the latter segment of the ipsilateral Willis circle, we could
WORLD NEUROSURGERY 126: 129-133, JUNE 2019
perform the direct ligation of the left ICA for the left EICA aneurysm of the patient. No neurologic events occurred following the ligation. In addition, concerning dealing with the disabling and fatal ischemic stroke in the central nervous system associated with EICA aneurysm, the end-to-end anastomosis following the resection of the EICA aneurysm should be an effective measure, and saphenous vein or prosthetic grafts also can be regarded as grafts of interposition.3 However, a previous study on EICA aneurysm indicated that open surgery, resection of EICA aneurysm, and anastomosis by prosthetic graft following the aforementioned resection may account for satisfied prognosis in the early- and long-term effect, but postoperative injury of cranial nerve should not be ignored.8 Unlike patients with cardiovascular diseases benefiting from the antiplatelet or anticoagulant measurements, the victims of EICA aneurysm cannot achieve similar beneficial effects.2 Therefore, no antiplatelet or anticoagulant measurements were performed before ligation of the left ICA and the ipsilateral STA-MCA bypass in the present study. Preoperative symptoms, DSA, CTP, and MRI revealed no indication of HMMD. In other words, the patient suffers HMMD (or HMMD transformation of ischemic MMD) and the concomitant EICA aneurysm. The standardized guidelines concerning HMMD and the concomitant EICA aneurysm are lacking and controversial for rare cases. Usually, if a patient suffers EICA aneurysm and intracranial cerebrovascular lesions, neurosurgeons should take careful consideration.9 Ge et al.10 investigated and indicated that compared with conservative treatment, surgical revascularization including STA-MCA bypass was superior in decreasing the recurrence of ischemic stroke and hemorrhage, regardless of mortality. However, collateral circulation is a major factor and should be analyzed carefully preoperatively.11 What is more, we performed the ipsilateral STA-MCA bypass for the concurrent MMD to prevent stroke. What is the reason for hemorrhagic stroke following the ligation of EICA and the left STA-MCA bypass in this present patient with EICA aneurysm and MMD? Some authors
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CASE REPORT QINGDONG HAN ET AL.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 3. Computed tomography (CT) shows hemorrhagic stroke in the right basal ganglia region 20 days after left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass (A). The hematoma disappeared 20 days after the onset of the hemorrhagic stroke (B). Right STA-MCA bypass was carried out 6 months after the left STA-MCA bypass
insist that hemorrhage of MMD is attributed to the aneurismal rupture of the collateral vessel located in the skull base, which may be the outcome of the dilation of the anterior choroidal artery and posterior communicating artery.10 Even cerebral hyperperfusion syndrome may be a culprit of postoperative hemorrhage following STA-MCA bypass in patients with MMD.12 However, surgical bypass has uncertain safety and efficacy.13 When it comes to the reason of cerebral hemorrhage happening on the contralateral side after a reconstruction operation to MMD, the change in hemodynamics is an important factor. Our patient had no history of diabetes, hypertension, alcohol abuse, and smoking. We found no evidence of hypertension in the perioperative phase, and the data of cerebral blood flow and radiography illustrated no signs of cerebral hyperperfusion syndrome following the ligation and bypass. Whether the cerebral dynamic changes before and after the
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(C). CT angiography, digital subtraction angiography, and CT perfusion (thick arrows) indicate the efficacy of the bilateral STA-MCA bypass (CeF). The disappearance of the left extracranial internal carotid aneurysm was verified 12 months following the ligation of extracranial internal carotid artery (thin arrows) (C, E).
ligation and bypass led to the hemorrhage still need further study.
verify safer and more effective treatment techniques.
CONCLUSIONS
REFERENCES
We present a rare case of simultaneous EICA aneurysm and HMMD. Due to the anastomoses between the front segment of the Willis circle and the latter segment of the Willis circle in the patient, ligation of the left EICA caused the disappearance of the EICA aneurysm with no neurologic abnormalities. Simultaneous STA-MCA bypass can be regarded as an effective choice for HMMD. However, further studies should be performed to confirm the effectiveness of surgical revascularization and simultaneous ligation of the EICA and STA-MCA bypass in a patient with EICA aneurysm and HMMD. Due to few cases and limited guidelines, neurosurgeons should pay more attention to the type of complicated lesions located in the EICA aneurysm and HMMD and summarize and
1. Claus I, Jacobs B, De Groote L, et al. Endovascular repair of an extracranial carotid artery aneurysm complicated by late stent thrombosis. Acta Chir Belg. 2018:1-5. https://doi.org/10.1080/00015458. 2018.1491515. 2. Pourier VEC, Welleweerd JC, Kappelle LJ, et al. Experience of a single center in the conservative approach of 20 consecutive cases of asymptomatic extracranial carotid artery aneurysms. Eur J Neurol. 2018;25:1285-1289. 3. Jiber H, Zrihni Y, Naouli H, et al. Fibrodysplasic aneurysms of the extracranial internal carotid artery: a new case report. Pan Afr Med J. 2017;28:170. 4. Domanin M, Lanfranconi S, Romagnoli S, et al. A rare cause of juvenile stroke: extracranial carotid artery aneurysm with venous complete reconstruction of the carotid bifurcation. Pediatr Neurosurg. 2018;53:275-279. 5. Ben Jamm H, Lagha A, Diope A, et al. Surgical management of internal carotid artery aneurysm near the skull base. J Med Vasc. 2018;43:262-266.
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.110
CASE REPORT QINGDONG HAN ET AL.
6. Guzhin VE, Dubovoy AV, Cherepanov AV. Surgical treatment of distal extracranial internal carotid artery aneurysms associated with pathological artery kinking. Zh Vopr Neirokhir Im N Burdenko. 2016;80:62-66 [in Russian]. 7. Schievink WI, Piepgras DG, McCaffrey TV, et al. Surgical treatment of extracranial internal carotid artery dissecting aneurysms. Neurosurgery. 1994;35: 809-815 [discussion: 815-816]. 8. Qi M, Ma Y, Jiao L. A hybrid operation for the treatment of extracranial carotid artery aneurysm with a 2-year follow-up [e-pub ahead of print]. Turk Neurosurg. 2018. https://doi.org/10.5137/10195149.JTN.21848-17.2. 9. van der Linde RA, Lind RC. Saccular aneurysm of the extracranial internal carotid artery. Eur J Vasc Endovasc Surg. 2018;55:850.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
10. Ge P, Zhang Q, Ye X, et al. Clinical features, surgical treatment, and long-term outcome in children with hemorrhagic moyamoya disease. J Stroke Cerebrovasc Dis. 2018;27:1517-1523. 11. Rodriguez-Hernandez A, Josephson SA, Lawton MT. Bypass surgery for the prevention of ischemic stroke: current indications and techniques. Neurocirugia (Astur). 2012;23:5-14. 12. Ishikawa T, Yamaguchi K, Kawashima A, et al. Predicting the occurrence of hemorrhagic cerebral hyperperfusion syndrome using regional cerebral blood flow after direct bypass surgery in patients with moyamoya disease. World Neurosurg. 2018;119: e750-e756. 13. Jang DK, Lee KS, Rha HK, et al. Bypass surgery versus medical treatment for symptomatic
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moyamoya disease in adults. J Neurosurg. 2017;127: 492-502.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 26 January 2019; accepted 10 February 2019 Citation: World Neurosurg. (2019) 126:129-133. https://doi.org/10.1016/j.wneu.2019.02.110 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
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Surgical Revascularization: Ligation of Extracranial Internal Carotid Artery and Superficial Temporal Artery-to-Middle Cerebral Artery Bypass in Patient with Extracranial Internal Carotid Aneurysm and Hemorrhagic Moyamoya Disease Qingdong Han, Peng Zhou, Yabo Huang
Key words Extracranial internal carotid artery aneurysm - Moyamoya disease - Surgical revascularization -
Abbreviations and Acronyms CT: Computed tomography CTA: Computed tomography angiography CTP: Computed tomography perfusion DSA: Digital subtraction angiography EICA: Extracranial internal carotid artery HMMD: Hemorrhagic moyamoya disease ICA: Internal carotid artery MMD: Moyamoya disease MRI: Magnetic resonance imaging STA-MCA: Superficial temporal artery-to-middle cerebral artery Department of Neurosurgery, the First Affiliated Hospital of Soochow University, Suzhou, Jiangsu, China To whom correspondence should be addressed: Yabo Huang, M.D. [E-mail: [email protected]] Qingdong Han and Peng Zhou contributed equally to this article are considered as coefirst authors. Citation: World Neurosurg. (2019) 126:129-133. https://doi.org/10.1016/j.wneu.2019.02.110
- BACKGROUND:
Extracranial internal carotid artery (EICA) aneurysm is regarded as a rare lesion. What is more, patients can suffer from EICA aneurysms and hemorrhagic moyamoya disease (MMD) simultaneously.
- CASE
DESCRIPTION: A 38-year-old man was admitted to our hospital and underwent emergent tracheal intubation for hoarseness and breathing difficulty for 1 day. He had a pulsating mass of 2.5 cm in diameter in the left cervical area. Imaging examinations revealed the left EICA aneurysm and MMD. The patient underwent left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass and ligation of the left EICA simultaneously. Computed tomography revealed hemorrhagic stroke in the right basal ganglia region 20 days after the bypass. The hematoma disappeared 20 days after the stroke. Right bypass was performed 6 months after the left bypass. The patient was discharged with no neurologic abnormalities 10 days after the right bypass. He lived a normal life, and no neurologic abnormalities were found at the 6-month follow-up. Imaging examinations indicated the efficacy of the bilateral STA-MCA bypass and verified the disappearance of the left EICA aneurysm after the ligation.
- CONCLUSIONS:
We present a case of EICA aneurysm and hemorrhagic MMD. Ligation of the left ECA eliminates the EICA aneurysm with no neurologic abnormalities. STA-MCA bypass can be regarded as an effective choice for hemorrhagic MMD. Further studies should be performed to confirm the effectiveness of combined surgical revascularization.
Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION Extracranial internal carotid artery (EICA) aneurysm is regarded as a rare lesion. EICA aneurysms account for less than 1% of all peripheral artery aneurysms.1 EICA aneurysm may lead to ischemic stroke, cranial nerve compression, and other severe effects.2 Limited guidelines can be used for the treatment of EICA aneurysms. What is more, if patients suffer EICA aneurysm and hemorrhagic moyamoya disease (HMMD) simultaneously, the complicated neck mass and intracranial lesions can make neurosurgeons feel overwhelmed and helpless because recent treatments concerning HMMD are lacking and, when existing, controversial.
We present a patient with EICA aneurysm and HMMD simultaneously who was treated with surgical revascularization of ligation of the EICA and superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass. To the best of our knowledge, the present study is the first report regarding EICA aneurysm and HMMD simultaneously treated with surgical revascularization of ligation of EICA and STA-MCA bypass. CASE DESCRIPTION The study received consent from the ethics committee of the First Affiliated of Soochow University. It also was approved by the patient and his family. A 38-year-old man was admitted to emergency department of our hospital and underwent emergent tracheal intubation for hoarseness and
WORLD NEUROSURGERY 126: 129-133, JUNE 2019
breathing difficulty for 1 day. Before the onset of his hoarseness, the patient had intermittent headache for more than 3 years. He had no history of diabetes, hypertension, cerebral stroke, head and neck injuries or tumors, alcohol abuse, and smoking. Findings of the physical examination indicated that he had a left cervical pulsating mass of 2.5 cm in diameter. Computed tomography (CT), computed tomography angiography (CTA), digital subtraction angiography (DSA), magnetic resonance imaging (MRI), and computed tomography perfusion (CTP) imaging of the head and neck revealed the EICA aneurysm (Figure 1AeG) and MMD (Figure 1GeK). CT, CTA, and DSA indicated the left EICA aneurysm (Figure 1AeG). DSA showed moyamoya disease (MMD) in the patient (Figure 1GeH). In addition, DSA showed
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CASE REPORT QINGDONG HAN ET AL.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 1. Computed tomography (CT), computed tomography angiography (CTA), digital subtraction angiography (DSA), magnetic resonance imaging (MRI), and computed tomography perfusion (CTP) imaging of extracranial internal carotid artery aneurysm (AeG) and hemorrhagic moyamoya disease (GeK). (AeG) CT, CTA, and DSA indicating left extracranial internal
the anastomoses of the posterior communicating artery (Figure 1I) between the frontal segments of the Willis circle and the latter segments of the Willis circle in the patient with MMD. No masses or ischemic lesions were found on CTP (Figure 1J) or MRI (Figure 1K). Two days after admission, the patient underwent left STA-MCA bypass and the ligation of the left EICA simultaneously in our Neurosurgery Department. Postoperative CTA and DSA indicated the validity of the left STA-MCA bypass in our patient (Figure 2AeC) and the disappearance of the left EICA aneurysm after the ligation (Figure 2A) 4 days following the operations, simultaneously (Figure 2AeC). No ischemic lesions were found on the postoperative CTP (Figure 2C) following the aforementioned operations. However, the patient suffered deteriorated muscle strength from 5 to 2 grades in the left limbs 20 days following the aforementioned operations. Emergency CT showed hemorrhagic stroke in the right basal ganglia region 10 days after the left STA-MCA bypass (Figure 3A). After medical treatment of 20 days, the hematoma disappeared 20 days after the onset of the
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carotid artery aneurysm. (GeH) DSA showing moyamoya disease in the patient. What’s more, the arrow in the DSA shows the anastomoses (I) between the front segment of the Willis circle and the latter segment of the Willis circle in the patient with moyamoya disease. No ischemic lesions could be found on CTP (J) and MRI (K).
hemorrhagic stroke (Figure 3B) and the muscle strength in the left limbs returned to 4 grades. Right STA-MCA bypass was carried out 6 months after the left STA-MCA bypass (Figure 3C). The patient was discharged with no neurologic abnormalities 10 days after the right STAMCA bypass. No antiplatelet or anticoagulant measurements were performed after the patient was discharged from hospital. Follow-up The patient lived a normal life and no neurologic abnormalities were found at the 6-month follow-up after being discharged. DSA and CTP indicated the efficacy of the bilateral STA-MCA bypass (Figure 3DeF) and verified the disappearance of the left ECIA aneurysm after the ligation. DISCUSSION The case of ECIA aneurysm and HMMD is complicated and rare. The percentage of ECIA aneurysms to all peripheral artery aneurysms is less than 1%.1 The majority of patients with ECIA aneurysms have no symptoms, but the rest may suffer ischemic stroke, cranial nerve
compression, pulsatile cervical mass leading to dyspnea or dysphagia, pain, or even fatal hematemesis or epistaxis, of which transient ischemic attack is well accepted as the most common symptom.3,4 However, the high rate of mortality and stroke associated with ECIA aneurysms usually is found in earlier research.2 In the present study, our patient suffered no transient ischemic attack but had cranial nerve compression, and an emergent tracheal intubation for hoarseness and breathing difficulty was performed. The mass effect on the cranial nerve could be the chief culprit. The etiology of ECIA aneurysm is diverse. Atherosclerosis, iatrogenic or accidental injuries, fibromuscular dysplasia, EhlerseDanlos syndrome, and Marfan syndrome could be responsible for the occurrence of ECIA aneurysm.4,5 Usually, ECIA aneurysms can be classified into 2 subgroups, true ECIAs and pseudoaneurysms: the former account for 37%e 42% of ECIA aneurysms for atherosclerosis, and the latter aneurysms are caused by trauma, radiotherapy in the treatment of neck cancer, and carotid endarectomy.1 Atherosclerotic ECIA aneurysms usually are located in the proximal internal
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.110
CASE REPORT QINGDONG HAN ET AL.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 2. Computed tomography angiography (CTA) and digital subtraction angiography (DSA) indicating the validity of the left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass in a patient with moyamoya disease and the disappearance of the left extracranial internal carotid aneurysm after the ligation simultaneously (AeC). The thick arrows on CTA (A) and DSA (B) show the bypass. The thin arrow (A) indicates the disappearance of the left extracranial internal carotid aneurysm after the ligation. No ischemic lesions could be found on computed tomography perfusion (C) following the left STA-MCA bypass and ligation of the extracranial internal carotid artery.
carotid artery (ICA) and its bifurcation, but dysplastic aneurysms are located in the distal segment of the ICA and related to chronic dissection.3 In the present study, the ECIA aneurysm was located in the proximal segment of the ICA. Before the onset of his hoarseness, the patient had intermittent headache for more than 3 years. He had no history of diabetes, hypertension, cerebral stroke, head and neck injuries, tumors, alcohol abuse, and smoking. Moreover, as to the intermittent headache, no specific reasons could be found. As a relatively young patient with EICA aneurysm, no various inflammatory diseases were found. Which measures we can take toward EICA aneurysms may be still be limited and disputable. Some researchers advise that whatever it takes, surgical measures should be taken.6 Open surgery, endovascular
treatment, or medical treatment for the lesions can be taken into consideration. A previous study showed that direct ligation of the EICA of patients suffering EICA aneurysms accounted for 20%e40% postoperative severe central nervous system stroke or high mortality.7 In other words, if the surgeons attach little importance to the collateral circulation of the Willis circle, direct ligation of the ICA may be lifethreatening. Even some authors performed high-flow extracranialeintracranial bypass to treat EICA aneurysm.6 Regarding preoperative risk evaluation, the cerebral blood perfusion by balloon occlusion test with DSA was carried out, and no abnormalities occurred in the patient. With good collateral circulation posterior communicating artery between the front segment and the latter segment of the ipsilateral Willis circle, we could
WORLD NEUROSURGERY 126: 129-133, JUNE 2019
perform the direct ligation of the left ICA for the left EICA aneurysm of the patient. No neurologic events occurred following the ligation. In addition, concerning dealing with the disabling and fatal ischemic stroke in the central nervous system associated with EICA aneurysm, the end-to-end anastomosis following the resection of the EICA aneurysm should be an effective measure, and saphenous vein or prosthetic grafts also can be regarded as grafts of interposition.3 However, a previous study on EICA aneurysm indicated that open surgery, resection of EICA aneurysm, and anastomosis by prosthetic graft following the aforementioned resection may account for satisfied prognosis in the early- and long-term effect, but postoperative injury of cranial nerve should not be ignored.8 Unlike patients with cardiovascular diseases benefiting from the antiplatelet or anticoagulant measurements, the victims of EICA aneurysm cannot achieve similar beneficial effects.2 Therefore, no antiplatelet or anticoagulant measurements were performed before ligation of the left ICA and the ipsilateral STA-MCA bypass in the present study. Preoperative symptoms, DSA, CTP, and MRI revealed no indication of HMMD. In other words, the patient suffers HMMD (or HMMD transformation of ischemic MMD) and the concomitant EICA aneurysm. The standardized guidelines concerning HMMD and the concomitant EICA aneurysm are lacking and controversial for rare cases. Usually, if a patient suffers EICA aneurysm and intracranial cerebrovascular lesions, neurosurgeons should take careful consideration.9 Ge et al.10 investigated and indicated that compared with conservative treatment, surgical revascularization including STA-MCA bypass was superior in decreasing the recurrence of ischemic stroke and hemorrhage, regardless of mortality. However, collateral circulation is a major factor and should be analyzed carefully preoperatively.11 What is more, we performed the ipsilateral STA-MCA bypass for the concurrent MMD to prevent stroke. What is the reason for hemorrhagic stroke following the ligation of EICA and the left STA-MCA bypass in this present patient with EICA aneurysm and MMD? Some authors
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CASE REPORT QINGDONG HAN ET AL.
REVASCULARIZATION OF EICA ANEURYSM AND HMMD
Figure 3. Computed tomography (CT) shows hemorrhagic stroke in the right basal ganglia region 20 days after left superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass (A). The hematoma disappeared 20 days after the onset of the hemorrhagic stroke (B). Right STA-MCA bypass was carried out 6 months after the left STA-MCA bypass
insist that hemorrhage of MMD is attributed to the aneurismal rupture of the collateral vessel located in the skull base, which may be the outcome of the dilation of the anterior choroidal artery and posterior communicating artery.10 Even cerebral hyperperfusion syndrome may be a culprit of postoperative hemorrhage following STA-MCA bypass in patients with MMD.12 However, surgical bypass has uncertain safety and efficacy.13 When it comes to the reason of cerebral hemorrhage happening on the contralateral side after a reconstruction operation to MMD, the change in hemodynamics is an important factor. Our patient had no history of diabetes, hypertension, alcohol abuse, and smoking. We found no evidence of hypertension in the perioperative phase, and the data of cerebral blood flow and radiography illustrated no signs of cerebral hyperperfusion syndrome following the ligation and bypass. Whether the cerebral dynamic changes before and after the
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(C). CT angiography, digital subtraction angiography, and CT perfusion (thick arrows) indicate the efficacy of the bilateral STA-MCA bypass (CeF). The disappearance of the left extracranial internal carotid aneurysm was verified 12 months following the ligation of extracranial internal carotid artery (thin arrows) (C, E).
ligation and bypass led to the hemorrhage still need further study.
verify safer and more effective treatment techniques.
CONCLUSIONS
REFERENCES
We present a rare case of simultaneous EICA aneurysm and HMMD. Due to the anastomoses between the front segment of the Willis circle and the latter segment of the Willis circle in the patient, ligation of the left EICA caused the disappearance of the EICA aneurysm with no neurologic abnormalities. Simultaneous STA-MCA bypass can be regarded as an effective choice for HMMD. However, further studies should be performed to confirm the effectiveness of surgical revascularization and simultaneous ligation of the EICA and STA-MCA bypass in a patient with EICA aneurysm and HMMD. Due to few cases and limited guidelines, neurosurgeons should pay more attention to the type of complicated lesions located in the EICA aneurysm and HMMD and summarize and
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REVASCULARIZATION OF EICA ANEURYSM AND HMMD
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Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 26 January 2019; accepted 10 February 2019 Citation: World Neurosurg. (2019) 126:129-133. https://doi.org/10.1016/j.wneu.2019.02.110 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
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