The Laryngoscope C 2015 The American Laryngological, V
Rhinological and Otological Society, Inc.
Case Report
Facial Palsy After Neck Arteriovenous Fistula Embolization Dong Hoon Lee, MD; Joon Kyoo Lee, MD; Tae Mi Yoon, MD; Sang Chul Lim, MD; Tae Sun Kim, MD Facial palsy after embolization of neck arteriovenous fistula is an extremely rare complication. In our case, complete facial palsy occurred after embolization and was successfully treated with superficial parotidectomy, vessel ligation, and plug removal. We report the first case of unusual facial palsy that developed 13 days after neck arteriovenous fistula embolization. As a result of our findings, we recommend, when a patient suffers from acute ipsilateral facial palsy after arteriovenous fistula embolization, the clinician should consider the possibility of complications of embolization, and immediate and appropriate management should be performed. Key Words: Facial palsy, arteriovenous fistula, embolization. Laryngoscope, 125:2125–2128, 2015
INTRODUCTION Embolization has already shown its efficacy in the treatment of spontaneous or posttraumatic lesions of the external carotid artery.1 Facial palsy after embolization is a rare complication.2–4 However, all of the previous cases occurred only with glomus tumors. To the best of our knowledge, a case of facial palsy after neck arteriovenous fistula embolization has not been reported in the literature. Herein, we report the first case of unusual facial palsy that developed 13 days after neck arteriovenous fistula embolization.
CASE REPORT A 39-year-old female presented with a right neck mass that had developed several decades ago. On physical examination, the patient had a soft, pulsatile mass in the right infra-auricular region. All cranial nerves were intact, and there were no neurological deficits. Computed tomography (CT) with enhancement revealed a huge fistula between the internal maxillary artery and the external jugular vein overlying the right mandibular ramus. One-stage angiography and super-selective preoperative embolization were scheduled. Following the preliminary, main-stem right external-carotid angiography, the two main feeding vessels—the internal maxil-
From the Department of Otolaryngology–Head and Neck Surgery (d.h.l., j.k.l., t.m.y., s.c.l.); and the Department of Neurosurgery (T.S.K.), Chonnam National University Medical School and Chonnam National University Hwasun Hospital, Hwasun, Republic of Korea. Send correspondence to Joon Kyoo Lee, MD, PhD, Department of Otolaryngology–Head and Neck Surgery, Chonnam National University Medical School and Hwasun Hospital, 160 Ilsimri, Hwasun, Jeonnam 519–809, Republic of Korea. E-mail: [email protected] Editor’s Note: This Manuscript was accepted for publication on December 22, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. DOI: 10.1002/lary.25151
Laryngoscope 125: September 2015
lary artery and the facial artery—were selectively catheterized and subsequently embolized with plug and coils. After embolization, the final check angiography demonstrated no residual fistula blush, and there was no neurological abnormality after the procedure. Physical examination showed decreased swelling in the neck. Thirteen days after embolization, the patient developed a total right-sided facial palsy of all branches (House-Brackmann grade V) (Fig. 1) with no other neurological deficits. It was initially thought that Bell’s palsy had occurred. In spite of steroid therapy, the facial palsy did not improve within 1 month. Follow-up CT with enhancement revealed marked dilatation and tortuosity of the right external jugular vein, which had compressed the main trunk of the facial nerve (Fig. 2). CT also showed continued occlusion of the right internal maxillary artery and the right facial artery. Surgery was performed at 71 days after development of facial palsy with a modified Blair incision. Superficial parotidectomy was carried out, and a huge retromandibular vein was encountered in the region of the main trunk of the facial nerve (Fig. 3). The retromandibular vein and the external jugular vein were ligated, and the plug was removed (Fig. 4). The right external carotid artery and its branches were ligated. The postoperative course was uneventful. Right facial palsy was still present at discharge but had improved (House-Brackmann grade III). At 3 months follow-up, the neck swelling had disappeared and the facial palsy had further improved (House-Brackmann grade II). By 21 months post-surgery, the facial palsy had completely recovered (Fig. 5).
DISCUSSION Embolization is a widely accepted method for the management of spontaneous or posttraumatic lesions of the external carotid artery.1 It is a simple, fast, and Lee et al.: Facial Palsy After Embolization
2125
Fig. 1. Preoperative facial photographs of the patient: (A) rest; (B) “e” phonation; (C) gentle eye closure; (D) forceful eye closure. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
reliable procedure, and it is the treatment of choice for arteriovenous fistula or false aneurysm as compared to a more complex surgery.5 Facial palsy after embolization is a rare complication; only four such cases have been reported.2–4 The mechanism of facial nerve palsy after embolization cannot be established with absolute certainty. We speculated that the delayed onset of facial nerve palsy may be due to several mechanisms. First, it could be due
to direct compression by the arteriovenous fistula, plug, and coils.6 The compression by plug and coils may occur gradually over a long-term period, causing direct damage or ischemia. Second, it could be explained by thrombus formation.6,7 The arteriovenous fistula, plug, and coils can lead to thrombophilia, which increases the subsequent risk of thrombosis. Third, this complication might occur as a result of axonotmetic injuries caused by traction of the microcatheter on the nerve during
Fig. 2. Computed tomography (CT) with enhancement reveals marked dilatation and tortuosity of the right external jugular vein (asterisk) and a previous embolized plug (arrow).
Fig. 3. The operative field after superficial parotidectomy shows a huge retromandibular vein and a previous embolized plug (asterisk). These have compressed the main trunk of the facial nerve (arrow). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
Laryngoscope 125: September 2015
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Lee et al.: Facial Palsy After Embolization
enced a delay (13 days) between the embolization and the facial palsy. In previous reported cases,3,4 facial palsy occurred within a few hours. This is why we initially thought it was Bell’s palsy, not a complication of embolization. However, we made an error with regard to appropriate management. When facial palsy did not improve after steroid treatment, we performed radiological examination and surgical management very late. If we had performed surgical treatment earlier, the recovery of facial palsy would have been better than at present. Fourth, facial function recovered after superficial parotidectomy, retromandibular and external jugular vein ligation, external carotid artery ligation, and plug removal, not after facial nerve decompression with a perineural incision. Fig. 4. The operative field after cut of the retromandibular vein shows a previous embolized plug (asterisk). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
CONCLUSION
embolization.8 Fourth, the possibility of facial nerve palsy related to direct toxic effects of dimethyl sulfoxide has also been suggested.8,9 However, it is difficult to explain the mechanism of delayed facial nerve palsy. It is very possible that delayed facial nerve palsy occurred due to multiple reasons. This case had some unique characteristics. First, the patient had a spontaneous intraparotid arteriovenous fistula, not a glomus tumor. Second, the main trunk of the facial nerve emerging from the stylomastoid foramen was the cause of facial palsy, not the mastoid segment of the facial nerve. Third, the patient experi-
BIBLIOGRAPHY
Facial palsy after neck arteriovenous fistula embolization is an extremely rare complication. However, awareness about this condition is necessary for effective management. When a patient suffers from acute ipsilateral facial palsy after arteriovenous fistula embolization, the clinician should consider the possibility of complications of embolization, and immediate and appropriate management should be provided.
1. Reizine D, Merland JJ, Birkui P, Leban M, Riche MC. Treatment of spontaneous intraparotid direct arteriovenous fistulae using a detachable balloon technique. J Neuroradiol 1985;12:35–43. 2. Valavanis A. Preoperative embolization of the head and neck: indications, patient selection, goals and precautions. AJNR Am J Neuroradiol 1986; 7:943–952. 3. Herdman RC, Gillespie JE, Ramsden RT. Facial palsy after glomus tumour embolization. J Laryngol Otol 1993;107:963–966.
Fig. 5. Facial photographs of the patient 21 months postoperatively: (A) rest; (B) “e” phonation; (C) gentle eye closure; (D) forceful eye closure. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
Laryngoscope 125: September 2015
Lee et al.: Facial Palsy After Embolization
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4. Marangos N, Schumacher M. Facial palsy after glomus jugulare tumour embolization. J Laryngol Otol 1999;131:268–270. 5. Bartoli JM, Triglia JM, Farnarier P, Moulin G, Kasbarian M. Embolization of an intraparotid false aneurysm of the external carotid artery: case report. Cardiovasc Intervent Radiol 1991;14:173–174. 6. Kashiwazaki D, Kuwayama N, Akioka N, Kuroda S. Delayed abducens nerve palsy after transvenous coil embolization for cavernous sinus dural arteriovenous fistulae. Acta Neurochir (Wien) 2014;156:97–101. 7. Nishino K, Ito Y, Hasegawa H, Kikuchi B, Shimbo J, Kitazawa K, Fujii Y. Cranial nerve palsy following transvenous embolization for a cavernous
Laryngoscope 125: September 2015
2128
sinus dural arteriovenous fistula: association with the volume and location of detachable coils. J Neurosurg 2008;109:208–214. 8. Nyberg EM, Chaudry MI, Turk AS, Turner RD. Transient cranial neuropathies as sequelae of Onyx embolization of arteriovenous shunt lesions near the skull base: possible axonotmetic traction injuries. J Neurointervent Surg 2013;5:e21. 9. Pei W, Huai-Zhang S, Shan-Cai X, Cheng G, Di Z. Isolated hypoglossal nerve palsy due to endovascular treatment of a dural arteriovenous fistula with Onyx-18. Interv Neuroradiol 2010;16:286– 289.
Lee et al.: Facial Palsy After Embolization
Rhinological and Otological Society, Inc.
Case Report
Facial Palsy After Neck Arteriovenous Fistula Embolization Dong Hoon Lee, MD; Joon Kyoo Lee, MD; Tae Mi Yoon, MD; Sang Chul Lim, MD; Tae Sun Kim, MD Facial palsy after embolization of neck arteriovenous fistula is an extremely rare complication. In our case, complete facial palsy occurred after embolization and was successfully treated with superficial parotidectomy, vessel ligation, and plug removal. We report the first case of unusual facial palsy that developed 13 days after neck arteriovenous fistula embolization. As a result of our findings, we recommend, when a patient suffers from acute ipsilateral facial palsy after arteriovenous fistula embolization, the clinician should consider the possibility of complications of embolization, and immediate and appropriate management should be performed. Key Words: Facial palsy, arteriovenous fistula, embolization. Laryngoscope, 125:2125–2128, 2015
INTRODUCTION Embolization has already shown its efficacy in the treatment of spontaneous or posttraumatic lesions of the external carotid artery.1 Facial palsy after embolization is a rare complication.2–4 However, all of the previous cases occurred only with glomus tumors. To the best of our knowledge, a case of facial palsy after neck arteriovenous fistula embolization has not been reported in the literature. Herein, we report the first case of unusual facial palsy that developed 13 days after neck arteriovenous fistula embolization.
CASE REPORT A 39-year-old female presented with a right neck mass that had developed several decades ago. On physical examination, the patient had a soft, pulsatile mass in the right infra-auricular region. All cranial nerves were intact, and there were no neurological deficits. Computed tomography (CT) with enhancement revealed a huge fistula between the internal maxillary artery and the external jugular vein overlying the right mandibular ramus. One-stage angiography and super-selective preoperative embolization were scheduled. Following the preliminary, main-stem right external-carotid angiography, the two main feeding vessels—the internal maxil-
From the Department of Otolaryngology–Head and Neck Surgery (d.h.l., j.k.l., t.m.y., s.c.l.); and the Department of Neurosurgery (T.S.K.), Chonnam National University Medical School and Chonnam National University Hwasun Hospital, Hwasun, Republic of Korea. Send correspondence to Joon Kyoo Lee, MD, PhD, Department of Otolaryngology–Head and Neck Surgery, Chonnam National University Medical School and Hwasun Hospital, 160 Ilsimri, Hwasun, Jeonnam 519–809, Republic of Korea. E-mail: [email protected] Editor’s Note: This Manuscript was accepted for publication on December 22, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. DOI: 10.1002/lary.25151
Laryngoscope 125: September 2015
lary artery and the facial artery—were selectively catheterized and subsequently embolized with plug and coils. After embolization, the final check angiography demonstrated no residual fistula blush, and there was no neurological abnormality after the procedure. Physical examination showed decreased swelling in the neck. Thirteen days after embolization, the patient developed a total right-sided facial palsy of all branches (House-Brackmann grade V) (Fig. 1) with no other neurological deficits. It was initially thought that Bell’s palsy had occurred. In spite of steroid therapy, the facial palsy did not improve within 1 month. Follow-up CT with enhancement revealed marked dilatation and tortuosity of the right external jugular vein, which had compressed the main trunk of the facial nerve (Fig. 2). CT also showed continued occlusion of the right internal maxillary artery and the right facial artery. Surgery was performed at 71 days after development of facial palsy with a modified Blair incision. Superficial parotidectomy was carried out, and a huge retromandibular vein was encountered in the region of the main trunk of the facial nerve (Fig. 3). The retromandibular vein and the external jugular vein were ligated, and the plug was removed (Fig. 4). The right external carotid artery and its branches were ligated. The postoperative course was uneventful. Right facial palsy was still present at discharge but had improved (House-Brackmann grade III). At 3 months follow-up, the neck swelling had disappeared and the facial palsy had further improved (House-Brackmann grade II). By 21 months post-surgery, the facial palsy had completely recovered (Fig. 5).
DISCUSSION Embolization is a widely accepted method for the management of spontaneous or posttraumatic lesions of the external carotid artery.1 It is a simple, fast, and Lee et al.: Facial Palsy After Embolization
2125
Fig. 1. Preoperative facial photographs of the patient: (A) rest; (B) “e” phonation; (C) gentle eye closure; (D) forceful eye closure. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
reliable procedure, and it is the treatment of choice for arteriovenous fistula or false aneurysm as compared to a more complex surgery.5 Facial palsy after embolization is a rare complication; only four such cases have been reported.2–4 The mechanism of facial nerve palsy after embolization cannot be established with absolute certainty. We speculated that the delayed onset of facial nerve palsy may be due to several mechanisms. First, it could be due
to direct compression by the arteriovenous fistula, plug, and coils.6 The compression by plug and coils may occur gradually over a long-term period, causing direct damage or ischemia. Second, it could be explained by thrombus formation.6,7 The arteriovenous fistula, plug, and coils can lead to thrombophilia, which increases the subsequent risk of thrombosis. Third, this complication might occur as a result of axonotmetic injuries caused by traction of the microcatheter on the nerve during
Fig. 2. Computed tomography (CT) with enhancement reveals marked dilatation and tortuosity of the right external jugular vein (asterisk) and a previous embolized plug (arrow).
Fig. 3. The operative field after superficial parotidectomy shows a huge retromandibular vein and a previous embolized plug (asterisk). These have compressed the main trunk of the facial nerve (arrow). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
Laryngoscope 125: September 2015
2126
Lee et al.: Facial Palsy After Embolization
enced a delay (13 days) between the embolization and the facial palsy. In previous reported cases,3,4 facial palsy occurred within a few hours. This is why we initially thought it was Bell’s palsy, not a complication of embolization. However, we made an error with regard to appropriate management. When facial palsy did not improve after steroid treatment, we performed radiological examination and surgical management very late. If we had performed surgical treatment earlier, the recovery of facial palsy would have been better than at present. Fourth, facial function recovered after superficial parotidectomy, retromandibular and external jugular vein ligation, external carotid artery ligation, and plug removal, not after facial nerve decompression with a perineural incision. Fig. 4. The operative field after cut of the retromandibular vein shows a previous embolized plug (asterisk). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
CONCLUSION
embolization.8 Fourth, the possibility of facial nerve palsy related to direct toxic effects of dimethyl sulfoxide has also been suggested.8,9 However, it is difficult to explain the mechanism of delayed facial nerve palsy. It is very possible that delayed facial nerve palsy occurred due to multiple reasons. This case had some unique characteristics. First, the patient had a spontaneous intraparotid arteriovenous fistula, not a glomus tumor. Second, the main trunk of the facial nerve emerging from the stylomastoid foramen was the cause of facial palsy, not the mastoid segment of the facial nerve. Third, the patient experi-
BIBLIOGRAPHY
Facial palsy after neck arteriovenous fistula embolization is an extremely rare complication. However, awareness about this condition is necessary for effective management. When a patient suffers from acute ipsilateral facial palsy after arteriovenous fistula embolization, the clinician should consider the possibility of complications of embolization, and immediate and appropriate management should be provided.
1. Reizine D, Merland JJ, Birkui P, Leban M, Riche MC. Treatment of spontaneous intraparotid direct arteriovenous fistulae using a detachable balloon technique. J Neuroradiol 1985;12:35–43. 2. Valavanis A. Preoperative embolization of the head and neck: indications, patient selection, goals and precautions. AJNR Am J Neuroradiol 1986; 7:943–952. 3. Herdman RC, Gillespie JE, Ramsden RT. Facial palsy after glomus tumour embolization. J Laryngol Otol 1993;107:963–966.
Fig. 5. Facial photographs of the patient 21 months postoperatively: (A) rest; (B) “e” phonation; (C) gentle eye closure; (D) forceful eye closure. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
Laryngoscope 125: September 2015
Lee et al.: Facial Palsy After Embolization
2127
4. Marangos N, Schumacher M. Facial palsy after glomus jugulare tumour embolization. J Laryngol Otol 1999;131:268–270. 5. Bartoli JM, Triglia JM, Farnarier P, Moulin G, Kasbarian M. Embolization of an intraparotid false aneurysm of the external carotid artery: case report. Cardiovasc Intervent Radiol 1991;14:173–174. 6. Kashiwazaki D, Kuwayama N, Akioka N, Kuroda S. Delayed abducens nerve palsy after transvenous coil embolization for cavernous sinus dural arteriovenous fistulae. Acta Neurochir (Wien) 2014;156:97–101. 7. Nishino K, Ito Y, Hasegawa H, Kikuchi B, Shimbo J, Kitazawa K, Fujii Y. Cranial nerve palsy following transvenous embolization for a cavernous
Laryngoscope 125: September 2015
2128
sinus dural arteriovenous fistula: association with the volume and location of detachable coils. J Neurosurg 2008;109:208–214. 8. Nyberg EM, Chaudry MI, Turk AS, Turner RD. Transient cranial neuropathies as sequelae of Onyx embolization of arteriovenous shunt lesions near the skull base: possible axonotmetic traction injuries. J Neurointervent Surg 2013;5:e21. 9. Pei W, Huai-Zhang S, Shan-Cai X, Cheng G, Di Z. Isolated hypoglossal nerve palsy due to endovascular treatment of a dural arteriovenous fistula with Onyx-18. Interv Neuroradiol 2010;16:286– 289.
Lee et al.: Facial Palsy After Embolization
