Journal of Clinical Neuroscience xxx (2018) xxx–xxx Contents lists available at ScienceDirect Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn Case report Internal carotid artery dissection without intracranial infarct following a minor shoulder trauma: The second pediatric case and review of the literature Dilek Cebeci a,⇑, Ebru Arhan a, Ercan Demir a, Murat Uçar b, Habibe Koç Uçar a, Aysße Serdaroğlu a, Zeynep Öztürk a a b Department of Pediatric Neurology, Gazi University Faculty of Medicine, 10th Floor Besevler, Ankara, Turkey Department of Radiology, Gazi University Faculty of Medicine, Ankara, Turkey a r t i c l e i n f o Article history: Received 1 April 2018 Accepted 8 July 2018 Available online xxxx Keywords: Carotid artery dissection Minor trauma Without infarct a b s t r a c t Introduction: Carotid artery dissections may occur in severe trauma such as motor vehicle accidents or may also develop due to minor trauma. We aimed to present a case with internal carotid artery dissection that referred to the pediatric neurology department due to speech impairment after minor shoulder trauma. Case: A previously healthy 10-year-old male patient was admitted to the pediatric emergency clinic due to headache, vomiting and speech impairment. In his story, we learned that he had bumped shoulder to shoulder with his friend about 6 h ago. He did not fall or hit his head. On his admission he could not speak and had right central facial paralysis. There was no infarct or diffusion limitation in MRI but MR angiography showed thinning in left internal carotid artery calibration. Fat-suppressed, non-contrast T1-weighted MRI showed that the left carotid artery had ring-shaped pathological signal changes. Low-molecular-weight heparin therapy was initiated with the diagnosis of carotid artery dissection (CAD). No hemiparesis or hemiplejia occurred in the follow-up of the patient. Within a few days, his speech improved. At the end of the first month, facial paralysis completely recovered. Conclusion: In carotid artery dissections, prodromal symptoms such as transient ischemic attack, like in our patient, are rarely present in children. For good long term outcomes, it is very important to suspect, diagnose and initiate appropriate treatment in a rapid manner in carotid artery dissection before severe neurological findings such as acute ischemic stroke develops. Ó 2018 Elsevier Ltd. All rights reserved. 1. Introduction Carotid artery dissections occur following a vessel wall tear or rupture of the vasa vasorum. It causes an intramural hematoma and a hemodynamically significant stenosis. Secondary thromboembolism in the vessel territory can cause an ischaemic stroke. Early recognition and adequate therapy are important to prevent or limit brain ischemia [1,2]. We present a case of internal carotid artery (ICA) dissection that occurred after a minor shoulder trauma and was detected before intracranial infarct developed. 2. Case report A previously healthy 10-year-old male patient was admitted to the pediatric emergency clinic due to headache, nausea, vomiting ⇑ Corresponding author. E-mail address: dilekcebeci@yahoo.com (D. Cebeci). and speech impairment. In his story, we learned that he had bumped shoulder to shoulder with his friend about 6 h ago. He did not fall or hit his head. The patient complained of headache and nausea after the collision. At first the patient had applied to another medical center and in the follow-up as he had swollen speech and could not speak, he was referred to our hospital. There was no flu symptoms, fever, diarrhea, syncope, seizure, motor loss in his anamnesis. He had no infection recently. He had no chronic illness and not using any medication. The parents were relatives. However, there was no neurological or rheumatologic disease in the family. The vital signs of the patient were normal. He could not speak, the cooperation was limited due to agitation. The right side of the mouth was immobil and the mouth was pulling to the left. He could close his eyes bilaterally. The further neurological examination was normal. The hemogram, blood gas and biochemical parameters of the patient were normal. Because of the trauma primarily intracranial hemorrhage was doubted and the brain CT was planned, yet it was normal. Because of dysphasia and central facial paralysis, brain MR and MR angiography were planned as it https://doi.org/10.1016/j.jocn.2018.07.012 0967-5868/Ó 2018 Elsevier Ltd. All rights reserved. Please cite this article in press as: Cebeci D et al. Internal carotid artery dissection without intracranial infarct following a minor shoulder trauma: The second pediatric case and review of the literature. J Clin Neurosci (2018), https://doi.org/10.1016/j.jocn.2018.07.012 2 D. Cebeci et al. / Journal of Clinical Neuroscience xxx (2018) xxx–xxx Fig. 1. (a) Diffusion MRI, normal. (b) MR angiography of the supra-aortic vessels, occlusion of the left ICA. (c, d) T1 fat-suppressed MRI, left carotid artery occlusion. might be acute ischemic stroke. There was no infarct or diffusion limitation in MRI but MR angiography showed that the left internal bifurcation was thin in the internal carotid artery calibration and some parts of internal carotid artery could not be visualized (Fig. 1). And also fat-suppressed, non-contrast T1-weighted MRI showed that the left carotid artery had ring-shaped pathological signal changes from the separation of the aortic arch to the cranial base. Low-molecular-weight heparin therapy was initiated with the diagnosis of carotid artery dissection (CAD). Thrombophilia panel, vasculitis markers, homocysteine and echocardiography were normal in terms of risk factors. No hemiparesis or hemiplejia occurred in the follow-up of the patient. Within a few days, his speech improved. At the end of the first month, facial paralysis completely recovered. 3. Discussion Arterial dissections of brain and neck are a major cause of cerebrovascular injuries in children and it’s seen in 20% of pediatric acute ischemic stroke [1] Dissections may be traumatic or spontaneous. In addition to severe traumas like motor vehicle accident, direct neck trauma; mild traumas like vomiting or coughing can also be seen (Table 1) [2–14]. Furthermore in roller coaster rides or water slide uses, sudden acceleration, deceleration, and rotation of the neck can stretch and compress ICA resulting in dissection. Moyamoya disease, inherited connective tissue disorders, arteriopathies, fibromuscular dysplasia, pharyngeal infections, atherosclerosis, cystic medial necrosis and infection can be the reasons of spontaneous or minor trauma dissections [2,15]. As it is in our case, in children carotid artery dissection (CAD) typically occurs in the distal cervical segment of the ICA before entering the carotid canal at the skull base. This is due to the immature cranio-cervical stability resulting from the weak neck musculature, more lax pediatric ligaments, large head to neck proportion and less protective reflexes [16,17]. The most common signs and symptoms in patients undergoing craniocervical artery dissection are hemiparesis, headache, aphasia, dysphasia, anopsia and an altered level of consciousness [3,18] (Table 1). In our case, dysphasia and facial palsy were present. Hemiparesia, which can result due to the progress of the dissection, didn’t develop. Spontaneous CADs are more commonly intracranial, whereas traumatic ones are more commonly extracranial [19]. In our case, post-traumatic extracranial dissection extending to the intracranial part of ICA was observed. However, as it is a minor trauma possible underlying diseases was investigated. Thrombophilia panel, echocardiography, vasculitis markers and homocysteine levels were normal. In pediatric CAD, prodromal symptoms rarely predict upcoming acute ischemic stroke (AIS). Usually, posttraumatic CAD is diagnosed after onset of focal neurological symptoms due to AIS. In Please cite this article in press as: Cebeci D et al. Internal carotid artery dissection without intracranial infarct following a minor shoulder trauma: The second pediatric case and review of the literature. J Clin Neurosci (2018), https://doi.org/10.1016/j.jocn.2018.07.012 3 D. Cebeci et al. / Journal of Clinical Neuroscience xxx (2018) xxx–xxx Table 1 Presenting symptoms, cause of injury and MRI or CT/MRA or CTA findings in pediatric cases. Study/year No. of patients Cause of injury Symptoms possibly linked with dissection Ischemic symptom MRI or CT/MRA or CTA Borges, 2000 2 Bar, 2002 Payton, 2004 1 1 Intraoral trauma Intraoral trauma Intraoral trauma ‘‘Laser tag” accident— bumped head Yes Yes Yes Yes Infarct/Right MCA Infarct/Left ICA Infarct/Left ICA Infarct/Left ICA Agner, 2005 Pierrot, 2006 2 2 Child abuse Fall Yes Yes Infarct/Left ICA Infarct/Right ICA Jariwala, 2006 1 Fall Motor vehicle accident No Yes No infarct/Right ICA Infarct/Right ICA Lin, 2007 1 Water slide injury Yes Infarct/Right ICA Levack, 2009 Moriarty, 2009 1 1 Shoulder belt Intraoral trauma Yes Yes Infarct/Left CCA Infarct/Left MCA Agostini, 2013 2 Yes Yes Infarct/Left ICA Infarct/Left MCA Nouh, 2015 Akbasß, 2016 Bent, 2016 1 1 1 Vigorous somersaults Violent head hyperextensionrotation episodes Roller coaster ride Water slide use Intraoral trauma Left hemiparesis, somnolence, confusion Left hemiparesis, aphasia Right hemiparesis, aphasia Slurred speech & headache; tongue deviated to right side and eyes deviated to left side, speech was dyarthric and pt was confused Seizures, left gaze unsteady gait, tremors right arm Decreased consciousness, left hemiplegia, central facial palsy Asymptomatic Increased confusion & lack of strength & sensation of entire left upper extremity; GCS Headache, vomiting, neck pain, numbness over left limbs, facial palsy, hemiplegia, & slurred speech Right hemiplegia & aphasia Decreased level of consciousness, rightsided Weakness Headache and visual trouble Irritability, right-sided weakness Yes Yes Yes Infarct/Right MCA Infarct/Left ICA Infarct/Right ICA Zant, 2017 Our patient 1 1 Minor head trauma Minor shoulder trauma Yes Yes Infarct/Right MCA and ICA No infarct/Left ICA Left-sided weakness and left facial drop Slurring of speech, right-sided weakness Diminished left extremity movement, dysconjugate gaze, fluctuating mental status Encephalopathy, left-sided hemiplegia Disphasia, facial palsy contrast to adult patients in which prodromal signs of upcoming AIS are frequently noted (transient ischemic attacks, amaurosis fugax, or local symptoms) children rarely present with these warning signs. Ideally, the diagnosis of CAD and the onset of therapy should occur during this latent period to prevent ischemic lesions and subsequently, improve the long-term outcome [1]. In our case, the patient had central facial paralysis and speech impairment. We defined it as transient ischemic attack, so we suspected of dissection and the diagnosis was confirmed by MRI. The treatment was initiated abruptly before intracranial infarct developed. In the diagnosis of CAD carotid artery ultrasonography, brain MR imaging, MR angiography, CT angiography, or conventional angiography may be used. Conventional angiography has been considered the diagnostic gold standard for CAD [1]. However conventional angiography has some disadvantages in children: the need for general anesthesia, difficulty and morbidity associated with obtaining arterial vascular access, the limiting use of ionizing radiation and the greater costs compared to MRI/MRA. MRI and MRA have also been shown to be as sensitive and specific as conventional angiography. Fat-suppressed, non-contrast T1-weighted MRI of both head and neck is diagnostic for intramural hematoma and dissection. Moreover, MRI allows to simultaneously rule out or detect acute ischemic stroke [1]. In our patient, as infarct or severe neurological deficits did not occur and also considering the complications, angiography was not performed. Fat-suppressed, noncontrast T1-weighted MRI showed that the left carotid artery had ring-shaped pathological signal changes from the separation of the aortic arch to the cranial base. In the pediatric population the treatment of CAD remains a controversial issue as controlled studies are lacking. Supportive measures such as hydration, control of hyperthermia, and seizures are complemented with the use of antiplatelet and anticoagulant medication. In adults, anticoagulation therapy is suggested for 3 to 6 months [20] to prevent injuries progressing to a higher injury grade, to reduce the number of strokes, and to prevent neurologic deterioration [20]. The use of antiplatelet therapy is even more controversial for patients who suffer a stroke following an intracranial dissection because of a real risk of intracranial hemorrhage [4]. Pandey et al. recommended 3 months of anticoagulation therapy and then change the regimen to antiplatelet therapy. We started anticoaguation, enoxaparin sodium in our case and planned to continue for 3 months. 4. Conclusion Besides the severe traumas, minor head and neck traumas may also cause carotid artery dissection. In contrast to adult patients in which prodromal signs of upcoming AIS like transient ischemic attacks are frequently noted, children rarely present with these warning signs [1]. 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