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	Type of Article: Case Report 

Hemorrhagic Paraganglioma of the Cauda Equina: 
Case Report and 
Review of the Magnetic Resonance Imaging Features
 
Woo Yat Ming, Peter, MBBS, MMedSc, FRCS (Edin)1
Hung Sze Lok, Remy, MBBS1
Wong Kai Sing, Alain, MBChB, FRCS (Edin)1
Iu Po Ping, MBBS, FRCR2
Chan Kwong Yau, MBChB, FRCS (Edin)1
Kwok Ching Kwong, John, MBBCh (NUI), FRCS (Glas), FRCS (Edin)1

1Department of Neurosurgery, Kwong Wah Hospital, Hong Kong
   2Department of Radiology, Kwong Wah Hospital, Hong Kong

Corresponding Author�s name and address: 
Woo Yat Ming, Peter 
Department of Neurosurgery,
Kwong Wah Hospital, 
25 Waterloo Road, Yau Ma Tei,
Hong Kong 
Phone: (+852) 3517 5052 
Fax: (+852) 3517 5260
Email: peterymwoo@gmail.com	

Running title: Hemorrhagic cauda equina paraganglioma: case report and MRI features. 

Conflicts of Interest: None declared.

Abstract 

Paragangliomas of the cauda equina are rare intradural spinal tumors known to be highly vascular lesions. Most patients are middle-age adult males that present with non-specific symptoms of chronic lumbar back pain. We report a patient with acute cauda equina syndrome diagnosed to have a hemorrhagic cauda equina paraganglioma. Intraoperatively a hypervascular intradural extramedullary tumor was encountered and completely excised with satisfactory functional recovery. Although there are no pathognomonic radiological signs several features supportive of a paraganglioma were identified on magnetic resonance imaging. We advocate meticulous imaging evaluation for distinguishing characteristics and introduce the �polar� sign as an additional suggestive feature for lesions reaching a length of two or vertebral bodies. Establishing a preoperative diagnosis is challenging, but is important in order to alert the surgeon of the operative risks.  

Keywords
Paraganglioma
Cauda equina
Magnetic resonance imaging 
INTRODUCTION

Paragangliomas of the cauda equina are rare accounting for 3.5% to 5% of all intradural spinal tumors of this region  ADDIN EN.CITE <EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1]. Although more than 210 cases have been documented only a few reports devoted to their radiological features exist in the literature  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1-10]. Regardless of their location paragangliomas are highly vascular  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2  HYPERLINK \l "_ENREF_11" \o "Demircivi Ozer, 2010 #33" 11]. We report a patient with a hemorrhagic paraganglioma that presented with acute cauda equina syndrome and review the magnetic resonance imaging (MRI) characteristics of these lesions. A radiological feature, the �polar� sign, is described to aid in the preoperative diagnosis and alert the neurosurgeon of the risk of encountering such vascular spinal tumors.

CASE REPORT

A 60 year-old male with a history of chronic lumbar back pain for three months experienced sudden lower limb weakness and retention of urine. Physical examination confirmed cauda equina syndrome characterized by paraplegia, lower limb hyporeflexia and lax anal tone. Pinprick sensation over the L4 to S1 dermatomes and proprioception were diminished bilaterally. Spinal MRI scans revealed a heterogeneously contrast-enhancing intradural extramedullary mass spanning from the T12 to L2 levels (Figure 1). On T2-weighted (T2W) imaging the lesion had a salt-and-pepper appearance with intralesional flow voids and a hypointense rim at its inferior border. In addition T2W hyperintense and T1-isointense signals within the superior and inferior poles of the lesion were noted. Neither cysts nor calcifications were observed. Axial imaging showed the lesion filled the spinal canal with no infiltration into the intervertebral foraminae. Pronounced serpentine perimedullary vascular flow void signals extending from the cervical to lumbar regions were also detected. Spinal catheter angiography showed no evidence of a vascular malformation or a tumor arterial capillary blush. The preoperative diagnosis was a conus medullaris ependymoma or a hemorrhagic Schwannoma.

A T12 to L2 multilevel laminectomy for tumor excision was performed. Intra-operatively the dura was highly vascular with engorged epidural veins. A highly vascular intradural extramedullary tan-colored tumor, with several arterial feeders and arterialized draining veins, was found adherent to the cauda equina. Gross total excision was performed with decompression of all nerve roots. No significant fluctuation in blood pressure was observed throughout the procedure. Histological examination revealed polygonal tumor cells with round nuclei arranged in compact nests, zellballen, surrounded by a delicate capillary network and spindle-shaped sustentacular cells (Figure 2). Immunohistochemically, the tumor cells were reactive to epithelial membrane antigen (EMA), neuron specific enolase (NSE) and synaptophysin. There was no expression of glial fibrillary acidic protein (GFAP). Sustentacular cells were positive for S-100 protein staining. The diagnosis of a cauda equina paraganglioma was confirmed. Post-operatively the patient experienced satisfactory neurological recovery and by six-months he could walk unaided without any sphincter dysfunction symptoms.DISCUSSION

The overall incidence of spinal paragangliomas in the general population is estimated to be 0.07% per 100 000  ADDIN EN.CITE <EndNote><Cite><Author>Yang</Author><Year>2005</Year><RecNum>52</RecNum><DisplayText>[2]</DisplayText><record><rec-number>52</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384745301">52</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Yang, S. Y.</author><author>Jin, Y. J.</author><author>Park, S. H.</author><author>Jahng, T. A.</author><author>Kim, H. J.</author><author>Chung, C. K.</author></authors></contributors><auth-address>Department of Neurosurgery, Seoul National University College of Medicine, 28 Yeongeon-dong, Seoul, Jongno-gu, Korea.</auth-address><titles><title>Paragangliomas in the cauda equina region: clinicopathoradiologic findings in four cases</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>49-55</pages><volume>72</volume><number>1</number><edition>2005/04/02</edition><keywords><keyword>Aged</keyword><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Diagnosis, Differential</keyword><keyword>Female</keyword><keyword>Humans</keyword><keyword>Magnetic Resonance Imaging</keyword><keyword>Male</keyword><keyword>Middle Aged</keyword><keyword>Neurilemmoma/pathology/surgery</keyword><keyword>Paraganglioma/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/*pathology/surgery</keyword><keyword>Treatment Outcome</keyword></keywords><dates><year>2005</year><pub-dates><date>Mar</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>15803375</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/15803375</url></related-urls></urls><electronic-resource-num>10.1007/s11060-004-2159-3</electronic-resource-num><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2]. With the advent of improved MRI resolution and precise immunohistochemical techniques this pathology is increasingly being recognized  ADDIN EN.CITE <EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1]. It remains unknown as to why the cauda equina is the most frequent central nervous system site with only sporadic reports of their occurrence in the cerebellum and pineal region. It has been postulated that aberrant migration of paraganglion cells along the neural tube transpires during embyrogenesis, but none have been identified in this region  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1  HYPERLINK \l "_ENREF_12" \o "Aghakhani, 1999 #1" 12]. Alternatively, the discovery of ectopic neuroblasts within the adult filum terminale and intradural nerve roots have prompted speculation that tumors could arise from dormant precursor cells  ADDIN EN.CITE <EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1]. 

Cauda equina paragangliomas usually affects adults of an age ranging from 12 to 71 years (mean, 48 years) with a slight male predominance (male: female, 1.54: 1)  ADDIN EN.CITE <EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1]. Spinal paragangliomas are slow-growing benign lesions that commonly manifest with non-specific chronic back pain associated with sciatica  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1  HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2]. Due to the difficulty in clinically distinguishing degenerative lumbar spondylosis from spinal paragangliomas, the diagnosis is often delayed until there is substantial tumor growth  ADDIN EN.CITE <EndNote><Cite><Author>Walsh</Author><Year>2005</Year><RecNum>48</RecNum><DisplayText>[13]</DisplayText><record><rec-number>48</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384745070">48</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Walsh, J. C.</author><author>O&apos;Brien, D. F.</author><author>Kumar, R.</author><author>Rawluk, D.</author></authors></contributors><auth-address>Department of Neurosurgery, Beaumont Hospital, Dublin, Ireland.</auth-address><titles><title>Paraganglioma of the cauda equina: a case report and literature review</title><secondary-title>Surgeon</secondary-title></titles><periodical><full-title>Surgeon</full-title></periodical><pages>113-6</pages><volume>3</volume><number>2</number><edition>2005/05/03</edition><keywords><keyword>Adult</keyword><keyword>*Cauda Equina</keyword><keyword>Humans</keyword><keyword>Male</keyword><keyword>Paraganglioma/*diagnosis/*surgery</keyword><keyword>Peripheral Nervous System Neoplasms/*diagnosis/*surgery</keyword></keywords><dates><year>2005</year><pub-dates><date>Apr</date></pub-dates></dates><isbn>1479-666X (Print)&#xD;1479-666X (Linking)</isbn><accession-num>15861947</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/15861947</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_13" \o "Walsh, 2005 #48" 13]. Occasionally patients present with cauda equina syndrome due to intratumoral hemorrhage and require early operative decompression  ADDIN EN.CITE <EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1]. Similar to other extra-adrenal paragangliomas, most cauda equina tumors are non-functional with only two reported patients exhibiting vasomotor amine syndrome  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_14" \o "Toyota, 1993 #47" 14  HYPERLINK \l "_ENREF_15" \o "Hong, 2012 #57" 15]. 

The diagnostic investigation of choice is MRI. Although there are no pathognomonic radiological signs, several features when considered in combination can be helpful. The differential diagnoses for intradural-extramedullary cauda equina region tumors include myxopapillary ependymomas (constituting 90% of lesions), Schwannomas, meningiomas, hemangioblastomas, epidermoid tumors and lipomas  ADDIN EN.CITE <EndNote><Cite><Author>Wein</Author><Year>2013</Year><RecNum>49</RecNum><DisplayText>[16]</DisplayText><record><rec-number>49</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384745150">49</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Wein, S.</author><author>Gaillard, F.</author></authors></contributors><auth-address>Department of Radiology, Royal Melbourne Hospital, Parkville, Victoria, Australia. sarawein@bigpond.com</auth-address><titles><title>Intradural spinal tumours and their mimics: a review of radiographic features</title><secondary-title>Postgrad Med J</secondary-title></titles><periodical><full-title>Postgrad Med J</full-title></periodical><pages>457-69</pages><volume>89</volume><number>1054</number><edition>2013/06/12</edition><dates><year>2013</year><pub-dates><date>Aug</date></pub-dates></dates><isbn>1469-0756 (Electronic)&#xD;0032-5473 (Linking)</isbn><accession-num>23749876</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/23749876</url></related-urls></urls><electronic-resource-num>10.1136/postgradmedj-2012-131503&#xD;postgradmedj-2012-131503 [pii]</electronic-resource-num><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_16" \o "Wein, 2013 #49" 16]. The latter two pathologies are readily excluded by virtue of their conspicuous MRI appearance. Epidermoid tumors and lipomas are hypointense and hyperintense respectively on T1W imaging displaying no contrast enhancement  ADDIN EN.CITE <EndNote><Cite><Author>Wein</Author><Year>2013</Year><RecNum>49</RecNum><DisplayText>[16]</DisplayText><record><rec-number>49</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384745150">49</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Wein, S.</author><author>Gaillard, F.</author></authors></contributors><auth-address>Department of Radiology, Royal Melbourne Hospital, Parkville, Victoria, Australia. sarawein@bigpond.com</auth-address><titles><title>Intradural spinal tumours and their mimics: a review of radiographic features</title><secondary-title>Postgrad Med J</secondary-title></titles><periodical><full-title>Postgrad Med J</full-title></periodical><pages>457-69</pages><volume>89</volume><number>1054</number><edition>2013/06/12</edition><dates><year>2013</year><pub-dates><date>Aug</date></pub-dates></dates><isbn>1469-0756 (Electronic)&#xD;0032-5473 (Linking)</isbn><accession-num>23749876</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/23749876</url></related-urls></urls><electronic-resource-num>10.1136/postgradmedj-2012-131503&#xD;postgradmedj-2012-131503 [pii]</electronic-resource-num><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_16" \o "Wein, 2013 #49" 16].

On MRI T1W sequences paragangliomas are predominantly isointense, centrally located and well circumscribed. Differentiating characteristics such as cystic degeneration are more commonly observed in Schwannomas and hemangioblastomas whereas calcifications are typical of meningiomas and ependymomas. The clinician should appreciate that paragangliomas are vascular tumors and identification of MRI features reflecting this quality is crucial  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1-11]. Paragangliomas invariably display intense gadolinium contrast enhancement, more so than ependymomas, and the pattern is often heterogeneous  ADDIN EN.CITE <EndNote><Cite><Author>Araki</Author><Year>1993</Year><RecNum>4</RecNum><DisplayText>[8]</DisplayText><record><rec-number>4</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744222">4</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Araki, Y.</author><author>Ishida, T.</author><author>Ootani, M.</author><author>Yamamoto, H.</author><author>Yamamoto, T.</author><author>Tsukaguchi, I.</author><author>Nakamura, H.</author></authors></contributors><auth-address>Department of Radiology, Osaka Rosai Hospital, Japan.</auth-address><titles><title>MRI of paraganglioma of the cauda equina</title><secondary-title>Neuroradiology</secondary-title></titles><periodical><full-title>Neuroradiology</full-title></periodical><pages>232-3</pages><volume>35</volume><number>3</number><edition>1993/01/01</edition><keywords><keyword>Cauda Equina/*pathology</keyword><keyword>Humans</keyword><keyword>*Magnetic Resonance Imaging</keyword><keyword>Male</keyword><keyword>Middle Aged</keyword><keyword>Paraganglioma/*diagnosis</keyword><keyword>Peripheral Nervous System Neoplasms/*diagnosis</keyword></keywords><dates><year>1993</year></dates><isbn>0028-3940 (Print)&#xD;0028-3940 (Linking)</isbn><accession-num>8459930</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/8459930</url></related-urls></urls><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_8" \o "Araki, 1993 #4" 8]. This is contrary to meningiomas and Schwannomas where homogenous enhancement is typical. These pathologies possess unique growth patterns with meningiomas exhibiting the dural tail sign and Schwannomas have a proclivity for intervertebral foramen involvement. Intratumoral flow-voids and a salt-and-pepper appearance best demonstrated by T2W imaging is supportive of paraganglioma vascularity  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1  HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2  HYPERLINK \l "_ENREF_9" \o "Taira, 2000 #46" 9  HYPERLINK \l "_ENREF_16" \o "Wein, 2013 #49" 16]. The presence of a hypointense rim on T2W sequences, known as the �cap� sign, a result of the paramagnetic effects of hemosiderin deposition is an additional hallmark  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1-10  HYPERLINK \l "_ENREF_17" \o "Gelabert-Gonzalez, 2005 #55" 17]. Congested serpentine perimedullary vessels are redolent of paragangliomas although hemangioblastomas also share this feature  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2  HYPERLINK \l "_ENREF_7" \o "Levy, 1993 #30" 7  HYPERLINK \l "_ENREF_9" \o "Taira, 2000 #46" 9  HYPERLINK \l "_ENREF_16" \o "Wein, 2013 #49" 16]. This is ascribed to either arteriovenous shunting within paragangliomas or venous congestion secondary to its mass effect  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1  HYPERLINK \l "_ENREF_2" \o "Yang, 2005 #52" 2  HYPERLINK \l "_ENREF_8" \o "Araki, 1993 #4" 8]. Ours is a distinctively rare case where such perimedullary vascular ectasia involved the entire spinal cord. 

To increase preoperative diagnostic accuracy we introduce the �polar� sign as a helpful radiological feature in differentiating other hemorrhagic cauda equina lesions such as ependymomas and hemangioblastomas. The sign comprises of hypointense signals on contrast T1W sequences and hyperintensities on T2W imaging within the lesion�s superior and/ or inferior poles representing hemorrhage at the tumor�s leading points of growth. The signal characteristics are in keeping with late subacute to chronic intratumoral hematomas that were confirmed by operative findings. The �polar� sign was also evident upon our retrospective review of MR images in previously published case reports that is hitherto described  ADDIN EN.CITE  ADDIN EN.CITE.DATA [ HYPERLINK \l "_ENREF_1" \o "Miliaras, 2003 #31" 1  HYPERLINK \l "_ENREF_4" \o "Boukobza, 1993 #7" 4  HYPERLINK \l "_ENREF_9" \o "Taira, 2000 #46" 9  HYPERLINK \l "_ENREF_12" \o "Aghakhani, 1999 #1" 12  HYPERLINK \l "_ENREF_13" \o "Walsh, 2005 #48" 13  HYPERLINK \l "_ENREF_17" \o "Gelabert-Gonzalez, 2005 #55" 17-19]. From this review this sign seems to be most apparent for spinal paragangliomas that have grown to length of two or more vertebral bodies.   

In the presence of ectactic spinal vessels associated with an intradural lesion catheter angiography is suggested. Not only to exclude arteriovenous malformations, but should a tumor vascular pedicle be identified, preoperative particle embolisation can assist in operative excision by reducing blood loss  ADDIN EN.CITE <EndNote><Cite><Author>Rodesch</Author><Year>2008</Year><RecNum>42</RecNum><DisplayText>[20]</DisplayText><record><rec-number>42</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744897">42</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Rodesch, G.</author><author>Gaillard, S.</author><author>Loiseau, H.</author><author>Brotchi, J.</author></authors></contributors><auth-address>Service de Neuroradiologie Diagnostique et Therapeutique, Hopital FOCH, 40 rue Worth, 92150, Suresnes, France. g.rodesch@hopital-foch.org</auth-address><titles><title>Embolization of intradural vascular spinal cord tumors : report of five cases and review of the literature</title><secondary-title>Neuroradiology</secondary-title></titles><periodical><full-title>Neuroradiology</full-title></periodical><pages>145-51</pages><volume>50</volume><number>2</number><edition>2007/10/13</edition><keywords><keyword>Adult</keyword><keyword>*Embolization, Therapeutic</keyword><keyword>Humans</keyword><keyword>Male</keyword><keyword>Middle Aged</keyword><keyword>Spinal Cord/*blood supply</keyword><keyword>Spinal Cord Neoplasms/*therapy</keyword><keyword>Vascular Neoplasms/*therapy</keyword></keywords><dates><year>2008</year><pub-dates><date>Feb</date></pub-dates></dates><isbn>0028-3940 (Print)&#xD;0028-3940 (Linking)</isbn><accession-num>17932665</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/17932665</url></related-urls></urls><electronic-resource-num>10.1007/s00234-007-0308-z</electronic-resource-num><language>eng</language></record></Cite></EndNote>[ HYPERLINK \l "_ENREF_20" \o "Rodesch, 2008 #42" 20].

CONCLUSION

Cauda equina paragangliomas are rare benign tumors with an excellent prognosis after complete resection. Meticulous MRI evaluation for the �polar� sign, �cap� sign and serpentine perimedullary vessels are important clues in supporting a preoperative radiological diagnosis. Awareness of their hypervascularity will aid the neurosurgeon in minimizing the risk of operative adverse events. 
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Figure 1. Sagittal MRI of the thoraco-lumbar spine showing an isointense intradural extramedullary spinal lesion extending from the T12 to the L2 levels (a, T1W).  Intense heterogeneous gadolinium contrast enhancement with intralesional superior and inferior polar hypointense signals, the �polar� sign (b, contrast T1W, white arrows). T2W imaging revealing corresponding hyperintense polar signal changes with a hypointense rim at the inferior aspect of the lesion, the �cap� sign (c, white arrowhead). Ectactic serpentine vascular flow voids rostral to the lesion involving the entire spinal cord (d, T2W, black arrowhead; e, T2W, black arrows). 

Figure 2. Photomicrographs of the cauda equina paraganglioma. Tumor parenchymal cells arranged in compact clusters (�zellballen�, a, H&E, x200, white arrow) and surrounded by a delicate capillary network. Polygonal parenchymal cells with round nuclei (b, H&E, x400, arrowhead) with supporting spindle-shaped sustentacular cells were observed (white arrow). Immunohistochemical staining showed strong parenchymal cell reactivity to synaptophysin (c) and sustentacular cell positivity to S-100 protein (d).    









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01�h2P��. ��A!�"�#��$��%��S����61�h2P:p�U���. ��A!�"�#��$��%������sD���y������K�	_ENREF_1f:D<EndNote><Cite><Author>Miliaras</Author><Year>2003</Year><RecNum>31</RecNum><DisplayText>[1-10]</DisplayText><record><rec-number>31</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744623">31</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Miliaras, G. C.</author><author>Kyritsis, A. P.</author><author>Polyzoidis, K. S.</author></authors></contributors><auth-address>Neurosurgical Institute, University of Ioannina, Department of Neurosurgery, Medical School, Ioannina, Greece. milasgrg@hotmail.com</auth-address><titles><title>Cauda equina paraganglioma: a review</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>177-90</pages><volume>65</volume><number>2</number><edition>2003/12/23</edition><keywords><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Humans</keyword><keyword>Paraganglioma/diagnosis/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/diagnosis/*pathology/surgery</keyword></keywords><dates><year>2003</year><pub-dates><date>Nov</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>14686739</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/14686739</url></related-urls></urls><language>eng</language></record></Cite><Cite><Author>Yang</Author><Year>2005</Year><RecNum>52</RecNum><record><rec-number>52</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384745301">52</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Yang, S. Y.</author><author>Jin, Y. J.</author><author>Park, S. H.</author><author>Jahng, T. A.</author><author>Kim, H. J.</author><author>Chung, C. K.</author></authors></contributors><auth-address>Department of Neurosurgery, Seoul National University College of Medicine, 28 Yeongeon-dong, Seoul, Jongno-gu, Korea.</auth-address><titles><title>Paragangliomas in the cauda equina region: clinicopathoradiologic findings in four cases</title><secondary-title>J Neurooncol</secondary-title></titles><periodical><full-title>J Neurooncol</full-title></periodical><pages>49-55</pages><volume>72</volume><number>1</number><edition>2005/04/02</edition><keywords><keyword>Aged</keyword><keyword>Cauda Equina/*pathology/surgery</keyword><keyword>Diagnosis, Differential</keyword><keyword>Female</keyword><keyword>Humans</keyword><keyword>Magnetic Resonance Imaging</keyword><keyword>Male</keyword><keyword>Middle Aged</keyword><keyword>Neurilemmoma/pathology/surgery</keyword><keyword>Paraganglioma/*pathology/surgery</keyword><keyword>Peripheral Nervous System Neoplasms/*pathology/surgery</keyword><keyword>Treatment Outcome</keyword></keywords><dates><year>2005</year><pub-dates><date>Mar</date></pub-dates></dates><isbn>0167-594X (Print)&#xD;0167-594X (Linking)</isbn><accession-num>15803375</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/15803375</url></related-urls></urls><electronic-resource-num>10.1007/s11060-004-2159-3</electronic-resource-num><language>eng</language></record></Cite><Cite><Author>Boncoeur-Martel</Author><Year>1996</Year><RecNum>41</RecNum><record><rec-number>41</rec-number><foreign-keys><key app="EN" db-id="92xstazzl225ete0ds9vz2dzadr9p90tvex2" timestamp="1384744837">41</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Boncoeur-Martel, M. P.</author><author>Lesort, A.</author><author>Moreau, J. J.</author><author>Labrousse, F.</author><author>Roche, I.</author><author>Bouillet, P.</author><author>Pascaud, J. L.</author><author>Dupuy, J. 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