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<article xmlns:xlink="http://www.w3.org/1999/xlink" xmlns:mml="http://www.w3.org/1998/Math/MathML" article-type="case-report"><?properties open_access?><front><journal-meta><journal-id journal-id-type="nlm-ta">J Cerebrovasc Endovasc Neurosurg</journal-id><journal-id journal-id-type="iso-abbrev">J Cerebrovasc Endovasc Neurosurg</journal-id><journal-id journal-id-type="publisher-id">JCEN</journal-id><journal-title-group><journal-title>Journal of Cerebrovascular and Endovascular Neurosurgery</journal-title></journal-title-group><issn pub-type="ppub">2234-8565</issn><issn pub-type="epub">2287-3139</issn><publisher><publisher-name>Korean Society of Cerebrovascular Surgeons; Society of Korean Endovascular Neurosurgeons</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">29152471</article-id><article-id pub-id-type="pmc">5678212</article-id><article-id pub-id-type="doi">10.7461/jcen.2017.19.2.111</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Internal Carotid Artery Dissection in Brazilian Jiu-Jitsu</article-title></title-group><contrib-group><contrib contrib-type="author" corresp="yes"><contrib-id contrib-id-type="orcid" authenticated="true">https://orcid.org/0000-0002-0683-5418</contrib-id><name><surname>Demartini</surname><given-names>Zeferino</given-names><suffix>Jr</suffix></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Rodrigues Freire</surname><given-names>Maxweyd</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Lages</surname><given-names>Roberto Oliver</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Francisco</surname><given-names>Alexandre Novicki</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Nanni</surname><given-names>Felipe</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Maranha Gatto</surname><given-names>Luana A.</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib><contrib contrib-type="author"><name><surname>Koppe</surname><given-names>Gelson Luis</given-names></name><xref ref-type="aff" rid="A1-jcen-19-111"/></contrib></contrib-group><aff id="A1-jcen-19-111">Department of Neurosurgery, Ponthific Catholic University of Paran&#xE1; - PUCPR - Curitiba (PR), Brazil.</aff><author-notes><corresp>Correspondence to Zeferino Demartini Jr. Hospital Universitario Cajuru - PUCPR, Departamento de Neurocirurgia, Avenida Sao Jose 300 - Cristo Rei, Curitiba- PR - Brazil 80.050-350. Tel: 55-41-3271-2801, Fax: 55-41-3271-3000, <email>demartiniz@gmail.com</email></corresp></author-notes><pub-date pub-type="ppub"><month>6</month><year>2017</year></pub-date><pub-date pub-type="epub"><day>30</day><month>6</month><year>2017</year></pub-date><volume>19</volume><issue>2</issue><fpage>111</fpage><lpage>116</lpage><history><date date-type="received"><day>28</day><month>2</month><year>2016</year></date><date date-type="rev-recd"><day>20</day><month>5</month><year>2016</year></date><date date-type="accepted"><day>29</day><month>9</month><year>2016</year></date></history><permissions><copyright-statement>&#xA9; 2017 Journal of Cerebrovascular and Endovascular Neurosurgery</copyright-statement><copyright-year>2017</copyright-year><copyright-holder>Journal of Cerebrovascular and Endovascular Neurosurgery</copyright-holder><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/"><license-p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">http://creativecommons.org/licenses/by-nc/3.0/</ext-link>) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Carotid artery dissection is a significant cause of stroke in young patients. It may be asymptomatic and go undiagnosed, or minimal transient manifestations may follow, commanding a higher index of suspicion than ordinarily exists to avoid misdiagnosis. Reported herein is a 27-year-old man who suffered extracranial internal carotid artery dissection while practicing a Brazilian Jiu-Jitsu submission maneuver. The patient's condition suddenly deteriorated one week later due to distal embolization and stroke. Despite endovascular treatment, with stenting of the cervical carotid artery, neurologic deficits remained. Of note, the objective in martial arts, which is to kill or incapacitate, has yet to be fully tempered in transitioning to sport. Brazilian Jiu-jitsu, a relatively new and fast-growing form of martial art, places emphasis on submission maneuvers. Related injuries are not common knowledge and are poorly described in the literature. This account is intended to shed light on the risk of this discipline. Through education and improved supervision, vascular injuries of this nature and the potentially lethal or disabling consequences may thus be prevented in young athletes.</p></abstract><kwd-group><kwd>Carotid artery injuries</kwd><kwd>Internal carotid artery dissection</kwd><kwd>Endovascular treatment</kwd><kwd>Vascular disorder</kwd><kwd>Sports</kwd><kwd>Martial arts</kwd></kwd-group></article-meta></front><body><sec sec-type="intro"><title>INTRODUCTION</title><p>Although carotid artery dissection is implicated in only 2.5% of all strokes,<xref rid="B3-jcen-19-111" ref-type="bibr">3)</xref> it is among the leading causes of stroke in patients &lt; 45 years old.<xref rid="B3-jcen-19-111" ref-type="bibr">3)</xref><xref rid="B5-jcen-19-111" ref-type="bibr">5)</xref><xref rid="B10-jcen-19-111" ref-type="bibr">10)</xref><xref rid="B23-jcen-19-111" ref-type="bibr">23)</xref> In terms of the various cervical vascular injuries chronicled in the literature that are due to blunt trauma, sports-related events are almost entirely confined to case reports or limited case series. Brazilian Jiu-Jitsu is a specific style of martial art that has gained popularity in recent years.<xref rid="B20-jcen-19-111" ref-type="bibr">20)</xref><xref rid="B21-jcen-19-111" ref-type="bibr">21)</xref> There is little actual documentation as yet, but the submission maneuvers that are practiced may predispose to certain injuries.<xref rid="B21-jcen-19-111" ref-type="bibr">21)</xref> This report describes a circumstance in which internal carotid artery dissection was directly attributable to a Brazilian jiu-jitsu maneuver.</p></sec><sec><title>CASE REPORT</title><p>A previously healthy 27 year-old professional male Brazilian jiu-jitsu fighter presented to the emergency room with headache, right motor deficit, and aphasia, all commencing 16 hours earlier. The patient had experienced a bout of severe neck pain one week earlier while practicing a submission maneuver known as the Rear Naked Choke or Lion Killer, with persistent pain locally thereafter. No family history of arterial dissection, stroke, connective tissue disorders, or migraine was elicited. The patient never smoked but the use of anabolic steroids (nandrolone and trenbolone) 1 month beforehand was reported.</p><p>Upon admission, findings included a Glasgow Coma Scale score of 11, right central facial paralysis, aphasia, and disproportionate right hemiparesis (muscular strength: right upper limb, grade 1/5; right lower limb, grade 3/5). There were no external signs of cervical trauma, such as bruising or abrasions.</p><p>Emergency computed tomography scan showed hypodensity in the left cerebral hemisphere (<xref ref-type="fig" rid="F1-jcen-19-111">Fig. 1A</xref>). Due to the nature of trauma sustained and a clinical suspicion of dissection, conventional cerebral angiography was performed on an emergency basis, using a Berenstein 5F diagnostic catheter (Merit Medical Systems Inc., South Jordan, UT, USA) over a Radiofocus Guide wire 0.035 inch &#xD7; 260 cm (Terumo Corp., Tokyo, Japan). Subsequently, decreased filling of the left middle cerebral artery, with several thrombotic occlusions of M2 and M3 segments (<xref ref-type="fig" rid="F2-jcen-19-111">Fig. 2A</xref>), and a dissection narrowing the origin of left internal carotid artery were observed (<xref ref-type="fig" rid="F2-jcen-19-111">Fig. 2B</xref>). The hydrophilic guidewire was positioned in the left external carotid artery, enabling replacement of the diagnostic catheter by an Epsylar 6F introducer sheath (OptiMed, Ettlingen, Germany), which was introduced into distal common carotid artery as a guiding catheter. A 5 mm Spider embolic protection device (ev3 [Covidien], Plymouth, MN, USA) was opened within left internal carotid artery, and angioplasty was done, deploying a Prot&#xE9;g&#xE9; RX self-expanding stent 6-8-40 mm (ev3 [Covidien]) to cover the entire length of the dissection (<xref ref-type="fig" rid="F2-jcen-19-111">Fig. 2C, D</xref>). The procedure was uneventful, using heparin for the duration, followed by acetylsalicylic acid (200 mg/day) and clopidogrel (75 mg/day) in the intensive care unit. Intracranial thrombolysis was not attempted.</p><p>During the first day of hospitalization, level of consciousness declined (Glasgow Coma Scale score, 8), calling for tracheal intubation. Another emergency computed tomography scan showed left hemispheric infarction, with hemorrhagic transformation and midline shift. An intraparenchymal catheter was implanted to monitor intracranial pressure and over the next 24 hours conservative measures were done, including mannitol, hypertonic saline infusion, induced hypothermia, pentobarbital coma therapy and moderate short term hyperventilation. However, despite all efforts, high intracranial pressure forced a decompressive craniectomy. On admission, routine laboratory testing for inflammatory, rheumatic, coagulation disorders had excluded other causes of ischemic stroke. Electrocardiography and transesophageal echocardiography showed no cardiac embolic source.</p><p>Severe pneumonia ultimately ensued, accompanied by extensive pleural effusions. Bilateral chest drainage was required, as well as eventual right pulmonary decortication. Nevertheless, the patient did recover (Glasgow Coma Scale score, 11) and was discharged after 3 months with a tracheostomy, gastrostomy, and persistence of prior deficits (modified Rankin Scale score, 4). A 2-day re-hospitalization was needed 5 months later for cranioplasty, inserting an acrylic plate (<xref ref-type="fig" rid="F1-jcen-19-111">Fig. 1B</xref>). One year later, physical therapy was still in progress (Glasgow Coma Scale score, 15; modified Rankin Scale score, 3). The patient walked with paretic gait due to right-sided weakness (muscular strength: right upper limb, grade 2/5; right lower limb, grade 4/5) and was dysphasic. He retired as a result of his handicaps.</p></sec><sec sec-type="discussion"><title>DISCUSSION</title><p>In the realm of martial arts, cervical vascular dissection typically involves the vertebral artery, having been reported with mixed martial arts,<xref rid="B20-jcen-19-111" ref-type="bibr">20)</xref> karate,<xref rid="B19-jcen-19-111" ref-type="bibr">19)</xref> wrestling,<xref rid="B4-jcen-19-111" ref-type="bibr">4)</xref> judo,<xref rid="B9-jcen-19-111" ref-type="bibr">9)</xref> kickboxing,<xref rid="B11-jcen-19-111" ref-type="bibr">11)</xref> and kung-fu.<xref rid="B17-jcen-19-111" ref-type="bibr">17)</xref> Although carotid artery dissection has also been described in taekwondo,<xref rid="B18-jcen-19-111" ref-type="bibr">18)</xref> karate<xref rid="B14-jcen-19-111" ref-type="bibr">14)</xref> and mixed martial arts,<xref rid="B13-jcen-19-111" ref-type="bibr">13)</xref> direct bodily impact (i.e., kicks and punches) may be responsible, which is not characteristic of Brazilian jiu-jitsu. As such, vulnerability of the extracranial carotid artery may reflect a tethering effect on the prepetrosal segment (at skull base entry), in conjunction with free neck mobility.<xref rid="B8-jcen-19-111" ref-type="bibr">8)</xref> On the other hand, this particular dissection involved the origin of internal carotid artery, suggesting the following biomechanical sequence: 1) carotid artery compression, creating a point of fixation and 2) subsequent hyperextension of the neck, with contralateral head rotation.<xref rid="B22-jcen-19-111" ref-type="bibr">22)</xref> The resultant traction would suffice to stretch and rupture the intimal layer of carotid artery distal to its point of fixation (<xref ref-type="fig" rid="F3-jcen-19-111">Fig. 3</xref>).</p><p>Carotid injury due to blunt cervical trauma may pose a diagnostic challenge. Symptoms are sometimes absent, minimal and transient, or even delayed for up to 12 months after the insult.<xref rid="B5-jcen-19-111" ref-type="bibr">5)</xref> The primary cause of stroke is embolic,<xref rid="B1-jcen-19-111" ref-type="bibr">1)</xref><xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref><xref rid="B3-jcen-19-111" ref-type="bibr">3)</xref> which explains the delay between injury and onset of neurologic deficits both here and in a similar incident with Taekwondo.<xref rid="B18-jcen-19-111" ref-type="bibr">18)</xref> Embolism likely results from localized thrombosis of disrupted endothelium.<xref rid="B12-jcen-19-111" ref-type="bibr">12)</xref></p><p>Despite inherent challenges, early diagnosis of cervical arterial dissection improves patient prognosis.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref> This patient scenario underscores the imperative for neurosurgeons and other sports physicians to consider arterial dissection when evaluating symptomatic athletes. Headache and neck pain are common, and pain may be the only indication.<xref rid="B8-jcen-19-111" ref-type="bibr">8)</xref> Other signs and symptoms are cranial nerve palsy, Horner's syndrome, pulsatile tinnitus, ataxia, vertigo, and dizziness.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref><xref rid="B4-jcen-19-111" ref-type="bibr">4)</xref> However, given the frequency of asymptomatic carotid stenosis, patients with risk factors who practice contact sports should be screened for this condition as well.<xref rid="B14-jcen-19-111" ref-type="bibr">14)</xref></p><p>A thorough history is fundamental, but because the diagnosis is confirmed through imaging, early studies may be prudent in this patient subset.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref><xref rid="B4-jcen-19-111" ref-type="bibr">4)</xref> Still, there is no consensus on imaging at present. Kerwin et al.<xref rid="B7-jcen-19-111" ref-type="bibr">7)</xref> advocate liberal screening of patients suffering blunt trauma and presenting with neurologic deficits, massive epistaxis, and cranial or spinal fractures. Doppler ultrasound and magnetic resonance or angiotomography are less invasive alternatives, whereas conventional angiography enables endovascular treatment and is thus preferable.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref><xref rid="B8-jcen-19-111" ref-type="bibr">8)</xref> Unfortunately, this athlete failed to seek help at the onset of pain, when a rapid and inexpensive carotid duplex scan may have prevented the life-threatening sequelae that were suffered.</p><p>Overall mortality in this setting is 3&#x2013;4%, with satisfactory outcomes in ~75% of survivors through conservative therapy.<xref rid="B7-jcen-19-111" ref-type="bibr">7)</xref> Prevention of stroke is the treatment goal; so despite the lack of randomized controlled trials, early anticoagulation is advised to halt the succession of new neurologic deficits.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref> However, evidence of symptomatic thromboembolic intracranial vascular occlusion and major stroke are contraindications for anticoagulation, according to Biondi et al.,<xref rid="B1-jcen-19-111" ref-type="bibr">1)</xref> posing a risk of hemorrhagic transformation. Stent-assisted endovascular repair was therefore warranted here, rather than anticoagulation. Emergency stenting also serves to stabilize the intimal flap and deter potential deterioration precipitated by emboli.<xref rid="B22-jcen-19-111" ref-type="bibr">22)</xref> Thrombolysis of intracerebral occlusions in an acute phase is also a safe and effective option, even following extracranial carotid artery stenting.<xref rid="B15-jcen-19-111" ref-type="bibr">15)</xref> It was contraindicated in this patient since he was beyond the 6-hour time window and had major involvement (over one-third) of the territory supplied by middle cerebral artery, heightening the risk of intracerebral hemorrhage.<xref rid="B6-jcen-19-111" ref-type="bibr">6)</xref></p><p>We acknowledge that no sport is considered completely safe, and the martial arts especially have evolved over millennia expressly as means to kill and disable.<xref rid="B16-jcen-19-111" ref-type="bibr">16)</xref> The transition to sport is a recent phenomenon that appears to be lacking in safety standards and regulations.<xref rid="B16-jcen-19-111" ref-type="bibr">16)</xref> Arterial dissection is a potentially devastating and underrecognized problem in these healthy young enthusiasts.<xref rid="B2-jcen-19-111" ref-type="bibr">2)</xref><xref rid="B4-jcen-19-111" ref-type="bibr">4)</xref><xref rid="B7-jcen-19-111" ref-type="bibr">7)</xref> Awareness of the risks must be increased and better supervision implemented to prohibit prolonged or overly vigorous moves.<xref rid="B12-jcen-19-111" ref-type="bibr">12)</xref><xref rid="B21-jcen-19-111" ref-type="bibr">21)</xref> Coaching and training staff must also caution participants against a sense of immunity, urging prompt medical attention for injuries sustained. 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tomography scan imaging: (A) at emergency admission, showing hypodensity of left cerebral hemisphere after &#x201C;windowing and leveling&#x201D; and (B) at postoperative cranioplasty (B).</title></caption><graphic xlink:href="jcen-19-111-g001"/></fig><fig id="F2-jcen-19-111" orientation="portrait" position="float"><label>Fig. 2</label><caption><title>Cerebral angiogram: (A) anteroposterior view of occluded M2 middle cerebral artery segments (arrows); (B) left common carotid artery in lateral view, showing narrowing at internal carotid artery origin (arrows); (C) stent placement; and (D) postoperative control images confirming adequate arterial patency, with mural compression of thrombus.</title></caption><graphic xlink:href="jcen-19-111-g002"/></fig><fig id="F3-jcen-19-111" orientation="portrait" position="float"><label>Fig. 3</label><caption><title>Artistic rendering of Brazilian Jiu-Jitsu maneuver known as Rear Naked Choke or Lion Killer: combined neck extension and head rotation (as a defense measure) stretches the compressed internal carotid artery at its origin, causing dissection.</title></caption><graphic xlink:href="jcen-19-111-g003"/></fig></floats-group></article>
