AMERI CAN JOURNAL OF OTOLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 90–9 2
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Facial nerve paralysis after pre-operative embolization of a paraganglioma☆,☆☆ Sameep Kadakia, MD⁎, Shira Koss, MD, Tova Fischer Isseroff, MD, Roy A. Holliday, MD, Ana H. Kim, MD Department of Otolaryngology-Head and Neck Surgery, New York Eye and Ear Infirmary-Mount Sinai Health System, 310 East 14th St, 6th Floor, New York, NY
ARTI CLE I NFO
A BS TRACT
Article history:
Vascular tumors pose a challenging problem in treatment, as surgical planning can be extensive.
Received 31 July 2014
Often times, pre-operative embolization is required to minimize blood loss during surgery. With the advent of new biochemical compounds, embolization modalities have evolved over the past decade. Although rare, side effects and complications of embolic materials have been cited sporadically in the literature. We present an interesting case of a patient afflicted with facial paralysis and other cranial neuropathies following embolization of a paraganglioma, along with the appropriate imaging that confirms the etiology of her paralysis. © 2015 Elsevier Inc. All rights reserved.
1.
Case presentation
A 48-year-old female with a past medical history significant only for asthma, presented to the clinic with multiple episodes of bloody otorrhea that resolved with packing. Physical exam showed a firm, nonpulsatile mass that completely filled the right canal to the meatus. Other than profound hearing loss, remaining lower cranial nerves were intact. Biopsy of the mass was consistent with jugulotympanic paraganglioma. CT and MRI imaging were performed to delineate the mass. Due to the lateral involvement of the canal with multiple episodes of bleeding requiring ER visits, decision was made for the patient to undergo surgical resection following pre-operative embolization. The patient was evaluated by the interventional radiology team and underwent embolization of the paraganglioma using a non-adhesive liquid embolic agent, ethylene vinyl alcohol copolymer dissolved in dimethyl sulfoxide and suspended in micronized tantalum powder (Onyx®, eV3 Inc). ☆
Immediately following the procedure, the patient awoke with a mild right facial paralysis, which progressed to complete paralysis the next morning. Patient also complained of right facial numbness and difficulty articulating. Flexible endoscopy revealed new onset of right true vocal fold paresis which one month prior was noted to be intact, surgery was canceled, and CT of the head without intravenous contrast was obtained (Figs. 1–4). What is your diagnosis?
2.
Discussion
2.1.
Diagnosis: iatrogenic cranial nerve neuropathy
The currently accepted treatment for paragangliomas of the head and neck is surgical extirpation. Owing to the vascular nature of these tumors, pre-operative embolization has been
There are no financial disclosures to declare. This article has not been published, nor is being considered for publication elsewhere. ⁎ Corresponding author at: Department of Otolaryngology-Head and Neck Surgery, New York Eye and Ear Infirmary-Mount Sinai Health System, 310 East 14th St, 6th Floor, New York, NY 10009. Tel.: +1 610 850 1884; fax: + 1 212 979 4315. E-mail addresses: [email protected] (S. Kadakia), [email protected] (S. Koss), [email protected] (T.F. Isseroff), [email protected] (R.A. Holliday), [email protected] (A.H. Kim). ☆☆
http://dx.doi.org/10.1016/j.amjoto.2014.08.014 0196-0709/© 2015 Elsevier Inc. All rights reserved.
AMERI CA N JOURNAL OF OT OLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 90–9 2
Fig. 1 – Non-contrast CT head, axial cut, showing embolic material linearly situated and extra embolic material at the anterior temporal canal junction. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
91
Fig. 3 – Non-contrast CT head, sagittal cut showing embolic material ear mass in the facial canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
performed to reduce blood loss intra-operatively and decrease operative time. Over the past years, many embolization materials have been used, each with varying risks and benefits. Cyanoacrylate glues rapidly solidify and permanently occlude small vessels, but provide poor occlusion of distal sites in the target feeding vessels [1]. Polyvinyl alcohol is used as solid particles that provide better distal occlusion than cyanoacrylate, but have resulted in only temporary occlusion of target vessels [2]. In 2005, ethylene vinyl alcohol dissolved in dimethyl sulfoxide was approved as a new embolic
material for the use of central nervous system vascular malformations [3]. Under the brand name Onyx® (eV3 Inc.), this material has been used off-label for pre-operative embolization of vascular tumors in the head and neck. Onyx® (eV3 Inc) is a non-adhesive liquid agent that combines the benefits of cyanoacrylate and polyvinyl alcohol in that it provides permanent occlusion of target vessels, allows for distal penetration, and has a longer solidification time [1]. Onyx® (eV3 Inc.), despite its benefits, has been implicated as the cause of post-embolic cranial neuropathies in a number of cases. Gartrell et al. in 2012 presented a review of 3 patients suffering from long term facial paralysis following Onyx® (eV3 Inc.) usage for pre-operative embolization of
Fig. 2 – Non-contrast CT head, sagittal cut, showing embolic material in the descending facial canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
Fig. 4 – Non-contrast CT head, coronal cut showing embolic material in the ear mass, including embolic material in the segment that seems to resemble horizontal semicircular canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
92
AMERI CAN JOURNAL OF OTOLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 90–9 2
glomus tumors. Two of these patients also suffered from other long-term cranial neuropathies as well, with none of the patients achieving full resolution [4]. These findings were corroborated by Gaynor et al. in 2014, showing that 2 out of 11 patients receiving Onyx® (eV3 Inc.) embolization developed permanent ipsilateral facial paralysis. Their findings were substantiated when the embolic material was found tracking along the vaso nervosum of the facial nerve intra-operatively [5]. In our case, the patient developed a facial nerve paralysis post-operatively, which progressed to become almost complete within 24 hours. Head CT showed hyperdense material tracking along facial nerve in various segments suggesting the presence of embolic polymer occluding the nerve. There was no evidence of stroke according to the CT scan. It is critical to understand the vascular supply to the facial nerve when considering post-embolization complications. The tympanic and mastoid portions of the nerve are supplied by the petrosal branch of the middle meningeal artery and the stylomastoid artery [6]. As the occipital artery is commonly catheterized during embolization, it is important to note that the stylomastoid artery arises from the occipital in the majority of patient (60%), but can also arise from the postauricular artery in 40% of individuals [7]. The literature also cites that 10% of people lack a middle meningeal contribution to the geniculate ganglion. The lack of a dual arterial supply to the geniculate ganglion predisposes these individuals to post-embolization facial nerve paralysis as the stylomastoid artery, which is often embolized, is the sole vascular supply in these patients for the tympanic and mastoid segments of the facial nerve [4,8]. The close anatomic relationship and shared blood supply between paragangliomas and cranial nerves may predispose patients to cranial nerve neuropathies following embolization. It is important to note that Onyx® (eV3 Inc) has numerous
advantages when compared to previous embolic adjuncts, but the risks should also be noted prior to use and may call for more thorough pre-embolization vascular mapping. To date, there are no studies comparing outcomes of Onyx® (eV3 inc.) with other embolic materials and therefore, conclusions are limited and based on sporadic cases. Practitioners should exercise caution when selecting the appropriate embolic agent, and patients should be notified of their potential risks.
REFERENCES
[1] Ayad M. Onyx: a unique neuroembolic agent. Expert Rev Med Devices 2006;3:705–15 [Pubmed: 17280535]. [2] Pauw BKH, Makek MS, Fisch U, et al. Preoperative embolization of paragangliomas (glomus tumors) of the head and neck: histopathologic and clinic features. Skull Base Surg 1993;3: 37–44 [Pubmed 17170888]. [3] Thiex R, Wu I, Mulliken JB, et al. Safety and clinical efficacy of Onyx for embolization of extracranial head and neck vascular anomalies. Am J Neuroradiol 2011;32:1082–6 [Pubmed: 21454409]. [4] Gartrell BC, Hansen MR, Gantz BJ, et al. Facial and lower cranial neuropathies after preoperative embolization of jugular foramen lesions with ethylene vinyl alcohol. Otol Neurotol 2012;33:1270–5. [5] Gaynor BG, Elhammady MS, Jethanamest D, et al. Incidence of cranial nerve palsy after preoperative embolization of glomus jugulare tumors using Onyx. J Neurosurg 2014;120:377–81. [6] Ozanne A, Pereira V, Krings T, et al. Arterial vascularization of the cranial nerves. Neuroimaging Clin N Am 2008;18: 431–9 [xii]. [7] Djindjian R. Super-selective arteriography of branches of the external carotid artery. Surg Neurol 1976;5:133–42. [8] Marangos N, Schumacher M. Facial palsy after glomus jugulare tumour embolization. J Laryngol Otol 1999;113: 268–70.
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Facial nerve paralysis after pre-operative embolization of a paraganglioma☆,☆☆ Sameep Kadakia, MD⁎, Shira Koss, MD, Tova Fischer Isseroff, MD, Roy A. Holliday, MD, Ana H. Kim, MD Department of Otolaryngology-Head and Neck Surgery, New York Eye and Ear Infirmary-Mount Sinai Health System, 310 East 14th St, 6th Floor, New York, NY
ARTI CLE I NFO
A BS TRACT
Article history:
Vascular tumors pose a challenging problem in treatment, as surgical planning can be extensive.
Received 31 July 2014
Often times, pre-operative embolization is required to minimize blood loss during surgery. With the advent of new biochemical compounds, embolization modalities have evolved over the past decade. Although rare, side effects and complications of embolic materials have been cited sporadically in the literature. We present an interesting case of a patient afflicted with facial paralysis and other cranial neuropathies following embolization of a paraganglioma, along with the appropriate imaging that confirms the etiology of her paralysis. © 2015 Elsevier Inc. All rights reserved.
1.
Case presentation
A 48-year-old female with a past medical history significant only for asthma, presented to the clinic with multiple episodes of bloody otorrhea that resolved with packing. Physical exam showed a firm, nonpulsatile mass that completely filled the right canal to the meatus. Other than profound hearing loss, remaining lower cranial nerves were intact. Biopsy of the mass was consistent with jugulotympanic paraganglioma. CT and MRI imaging were performed to delineate the mass. Due to the lateral involvement of the canal with multiple episodes of bleeding requiring ER visits, decision was made for the patient to undergo surgical resection following pre-operative embolization. The patient was evaluated by the interventional radiology team and underwent embolization of the paraganglioma using a non-adhesive liquid embolic agent, ethylene vinyl alcohol copolymer dissolved in dimethyl sulfoxide and suspended in micronized tantalum powder (Onyx®, eV3 Inc). ☆
Immediately following the procedure, the patient awoke with a mild right facial paralysis, which progressed to complete paralysis the next morning. Patient also complained of right facial numbness and difficulty articulating. Flexible endoscopy revealed new onset of right true vocal fold paresis which one month prior was noted to be intact, surgery was canceled, and CT of the head without intravenous contrast was obtained (Figs. 1–4). What is your diagnosis?
2.
Discussion
2.1.
Diagnosis: iatrogenic cranial nerve neuropathy
The currently accepted treatment for paragangliomas of the head and neck is surgical extirpation. Owing to the vascular nature of these tumors, pre-operative embolization has been
There are no financial disclosures to declare. This article has not been published, nor is being considered for publication elsewhere. ⁎ Corresponding author at: Department of Otolaryngology-Head and Neck Surgery, New York Eye and Ear Infirmary-Mount Sinai Health System, 310 East 14th St, 6th Floor, New York, NY 10009. Tel.: +1 610 850 1884; fax: + 1 212 979 4315. E-mail addresses: [email protected] (S. Kadakia), [email protected] (S. Koss), [email protected] (T.F. Isseroff), [email protected] (R.A. Holliday), [email protected] (A.H. Kim). ☆☆
http://dx.doi.org/10.1016/j.amjoto.2014.08.014 0196-0709/© 2015 Elsevier Inc. All rights reserved.
AMERI CA N JOURNAL OF OT OLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 90–9 2
Fig. 1 – Non-contrast CT head, axial cut, showing embolic material linearly situated and extra embolic material at the anterior temporal canal junction. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
91
Fig. 3 – Non-contrast CT head, sagittal cut showing embolic material ear mass in the facial canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
performed to reduce blood loss intra-operatively and decrease operative time. Over the past years, many embolization materials have been used, each with varying risks and benefits. Cyanoacrylate glues rapidly solidify and permanently occlude small vessels, but provide poor occlusion of distal sites in the target feeding vessels [1]. Polyvinyl alcohol is used as solid particles that provide better distal occlusion than cyanoacrylate, but have resulted in only temporary occlusion of target vessels [2]. In 2005, ethylene vinyl alcohol dissolved in dimethyl sulfoxide was approved as a new embolic
material for the use of central nervous system vascular malformations [3]. Under the brand name Onyx® (eV3 Inc.), this material has been used off-label for pre-operative embolization of vascular tumors in the head and neck. Onyx® (eV3 Inc) is a non-adhesive liquid agent that combines the benefits of cyanoacrylate and polyvinyl alcohol in that it provides permanent occlusion of target vessels, allows for distal penetration, and has a longer solidification time [1]. Onyx® (eV3 Inc.), despite its benefits, has been implicated as the cause of post-embolic cranial neuropathies in a number of cases. Gartrell et al. in 2012 presented a review of 3 patients suffering from long term facial paralysis following Onyx® (eV3 Inc.) usage for pre-operative embolization of
Fig. 2 – Non-contrast CT head, sagittal cut, showing embolic material in the descending facial canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
Fig. 4 – Non-contrast CT head, coronal cut showing embolic material in the ear mass, including embolic material in the segment that seems to resemble horizontal semicircular canal. Image provided courtesy of Dr. Roy Holliday, Director of Radiology at New York Eye and Ear Infirmary.
92
AMERI CAN JOURNAL OF OTOLAR YNGOLOGY–H E AD AN D N E CK M EDI CI N E AN D S U RGE RY 3 6 ( 2 0 15 ) 90–9 2
glomus tumors. Two of these patients also suffered from other long-term cranial neuropathies as well, with none of the patients achieving full resolution [4]. These findings were corroborated by Gaynor et al. in 2014, showing that 2 out of 11 patients receiving Onyx® (eV3 Inc.) embolization developed permanent ipsilateral facial paralysis. Their findings were substantiated when the embolic material was found tracking along the vaso nervosum of the facial nerve intra-operatively [5]. In our case, the patient developed a facial nerve paralysis post-operatively, which progressed to become almost complete within 24 hours. Head CT showed hyperdense material tracking along facial nerve in various segments suggesting the presence of embolic polymer occluding the nerve. There was no evidence of stroke according to the CT scan. It is critical to understand the vascular supply to the facial nerve when considering post-embolization complications. The tympanic and mastoid portions of the nerve are supplied by the petrosal branch of the middle meningeal artery and the stylomastoid artery [6]. As the occipital artery is commonly catheterized during embolization, it is important to note that the stylomastoid artery arises from the occipital in the majority of patient (60%), but can also arise from the postauricular artery in 40% of individuals [7]. The literature also cites that 10% of people lack a middle meningeal contribution to the geniculate ganglion. The lack of a dual arterial supply to the geniculate ganglion predisposes these individuals to post-embolization facial nerve paralysis as the stylomastoid artery, which is often embolized, is the sole vascular supply in these patients for the tympanic and mastoid segments of the facial nerve [4,8]. The close anatomic relationship and shared blood supply between paragangliomas and cranial nerves may predispose patients to cranial nerve neuropathies following embolization. It is important to note that Onyx® (eV3 Inc) has numerous
advantages when compared to previous embolic adjuncts, but the risks should also be noted prior to use and may call for more thorough pre-embolization vascular mapping. To date, there are no studies comparing outcomes of Onyx® (eV3 inc.) with other embolic materials and therefore, conclusions are limited and based on sporadic cases. Practitioners should exercise caution when selecting the appropriate embolic agent, and patients should be notified of their potential risks.
REFERENCES
[1] Ayad M. Onyx: a unique neuroembolic agent. Expert Rev Med Devices 2006;3:705–15 [Pubmed: 17280535]. [2] Pauw BKH, Makek MS, Fisch U, et al. Preoperative embolization of paragangliomas (glomus tumors) of the head and neck: histopathologic and clinic features. Skull Base Surg 1993;3: 37–44 [Pubmed 17170888]. [3] Thiex R, Wu I, Mulliken JB, et al. Safety and clinical efficacy of Onyx for embolization of extracranial head and neck vascular anomalies. Am J Neuroradiol 2011;32:1082–6 [Pubmed: 21454409]. [4] Gartrell BC, Hansen MR, Gantz BJ, et al. Facial and lower cranial neuropathies after preoperative embolization of jugular foramen lesions with ethylene vinyl alcohol. Otol Neurotol 2012;33:1270–5. [5] Gaynor BG, Elhammady MS, Jethanamest D, et al. Incidence of cranial nerve palsy after preoperative embolization of glomus jugulare tumors using Onyx. J Neurosurg 2014;120:377–81. [6] Ozanne A, Pereira V, Krings T, et al. Arterial vascularization of the cranial nerves. Neuroimaging Clin N Am 2008;18: 431–9 [xii]. [7] Djindjian R. Super-selective arteriography of branches of the external carotid artery. Surg Neurol 1976;5:133–42. [8] Marangos N, Schumacher M. Facial palsy after glomus jugulare tumour embolization. J Laryngol Otol 1999;113: 268–70.
