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CASE REPORT
Year : 2020  |  Volume : 15  |  Issue : 4  |  Page : 1050-1054

Subarachnoid hemorrhage and internal carotid artery dissection and occlusion following self-enucleation


1 University of South Florida Morsani College of Medicine, Tampa, Florida, USA
2 Department of Neurosurgery, Institute for Brain Protection Sciences, Johns Hopkins All Children's Hospital, Johns Hopkins University School of Medicine, Saint Petersburg, Florida, USA
3 Department of Neurosurgery and Brain Repair, Tampa General Hospital, University of South Florida Morsani College of Medicine, Tampa, Florida, USA
4 Department of Ophthalmology, Tampa General Hospital, University of South Florida Morsani College of Medicine, Tampa, Florida, USA

Date of Submission28-Apr-2020
Date of Decision14-Jun-2020
Date of Acceptance10-Aug-2020
Date of Web Publication19-Oct-2020

Correspondence Address:
Dr. Siviero Agazzi
Department of Neurosurgery and Brain Repair, Tampa General Hospital, University of South Florida Morsani College of Medicine, Tampa, Florida
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ajns.AJNS_183_20

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  Abstract 

Self-enucleation is an uncommon type of major self-injury, which may lead to severe neurological deficits and life-threatening complications, such as subarachnoid hemorrhage (SAH) and internal carotid artery (ICA) dissection and occlusion. Our patient is a 53-year-old man with a history of bipolar disorder and schizophrenia who presented with SAH, intraventricular hemorrhage, ICA dissection and occlusion, and right cerebral infarct following self-enucleation. Despite a Glasgow Coma Score of 6 on initial presentation, he improved with conservative management. He achieved a near-complete neurological recovery, with residual left lower extremity weakness and mild confusion. Self-enucleation is a major neurologic, ophthalmologic, and psychiatric emergency with a potential for serious neurological complications and contralateral visual loss. Yet, conservative management may lead to dramatic recovery.

Keywords: Dissection, internal carotid artery, occlusion, self-enucleation, subarachnoid hemorrhage


How to cite this article:
Joud H, Noureldine MH, Peto I, Kumar JI, Bajric J, Agazzi S. Subarachnoid hemorrhage and internal carotid artery dissection and occlusion following self-enucleation. Asian J Neurosurg 2020;15:1050-4

How to cite this URL:
Joud H, Noureldine MH, Peto I, Kumar JI, Bajric J, Agazzi S. Subarachnoid hemorrhage and internal carotid artery dissection and occlusion following self-enucleation. Asian J Neurosurg [serial online] 2020 [cited 2021 Jan 26];15:1050-4. Available from: https://www.asianjns.org/text.asp?2020/15/4/1050/298554


  Introduction Top


Self-enucleation, also known as auto-enucleation or oedipism, is an uncommon type of self-mutilation that is classified as a major self-injury.[1],[2] It is more common in men than in women[3],[4] and is overwhelmingly associated with acute psychosis due to untreated psychiatric illnesses or drug use.[1],[4],[5],[6] Self-enucleation involves avulsion of the extraocular muscles, optic nerve, and ophthalmic artery (OA), which may lead to devastating neurovascular complications. These include contralateral visual field deficits due to optic chiasm injury, subarachnoid hemorrhage (SAH), cerebrospinal fluid leak, infection, and internal carotid artery (ICA) dissection.[4]

Only 10 cases of traumatic SAH due to OA avulsion have been reported in the literature,[4],[7],[8],[9] five of which were reported to be associated with diffuse hemorrhage in the basal cisterns, mimicking the pattern seen in aneurysmal SAH. Given the risk of cerebral vasospasm following SAH, which may lead to ischemic infarcts, these patients require careful monitoring in an intensive care unit.[5] To the best of our knowledge, only one case of ICA dissection after self-enucleation has previously been reported in the literature.[4] We present a unique case of self-enucleation with both SAH as well as ICA dissection and occlusion.


  Case Report Top


The patient is a 53-year-old male with a history of bipolar disorder, schizophrenia, and multiple suicidal attempts who electively stopped his psychiatric medications at 1 month prior to presentation. During an acute psychotic episode, the patient self-enucleated his right eye. He arrived unconscious at the emergency department (ED), and he was intubated. The empty right orbit was washed out and packed with iodoform gauze in the ED, and the patient was then transferred to our facility. Neurological examination revealed a Glasgow Coma Score (GCS) of 6, withdrawal in all extremities except left upper extremity, and positive left corneal, cough, and gag reflexes. Computed tomography (CT) images showed extensive SAH (Fisher grade 4), as well as intraventricular hemorrhage and acute hydrocephalus [Figure 1]. CT angiography and digital subtraction angiography (DSA) revealed occlusion of the right ICA [Figure 2]. The gauze in the right orbit was gently removed with irrigation and no bleeding was encountered. A plastic conformer was placed inside the eyelids to maintain the socket space and allow the socket to granulate in, and antibiotic eye ointment was applied to prevent infection.
Figure 1: (a) Axial computed tomography head image showing acute diffuse subarachnoid hemorrhage in the frontotemporoparietal and basilar cisterns. (b) Axial computed tomography head images more superiorly showing intraventricular hemorrhage in the 3rd ventricle

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Figure 2: (a) Coronal computed tomography angiography image demonstrating occlusion of the right internal carotid artery just distal to the bifurcation of the right common carotid artery. (b) Coronal three-dimensional reconstruction of the computed tomography angiography image in Figure 2a. (c) Lateral digital subtraction angiography with injection into right common carotid artery demonstrating occlusion of the right internal carotid artery near the bifurcation of the right carotid bifurcation

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Per discussion with the family, the patient was initially managed conservatively in the neurointensive care unit. His GCS, however, improved, and he was extubated on posttrauma day (PTD) 2, with a GCS of 13. Mental status then declined overnight. An external ventricular drain (EVD) was placed, revealing an elevated opening pressure of 30 cmH2O, and resulted in immediate improvement. Neurological examination showed a reactive left pupil and full motor strength in all extremities, except for weakness in the left distal lower extremity that persisted throughout the hospital stay. Magnetic resonance imaging (MRI) demonstrated acute ischemia in the right distal anterior cerebral artery (ACA) and middle cerebral artery (MCA) territories [Figure 3]. Repeat cerebral angiogram confirmed complete occlusion of the right cervical ICA (C1–C7 segments) with robust cross-filling of the right MCA from the anterior communicating and posterior communicating arteries [Figure 4]. Daily transcranial Doppler (TCD) readings showed elevated flow velocities in bilateral ACAs and MCAs. The patient failed multiple EVD clamping trials and had persistent hydrocephalus, necessitating placement of a ventriculoperitoneal shunt. Head CT on PTD 28 showed a hypodensity within the right supplementary motor area [Figure 5], consistent with subacute ischemia seen on prior MRI studies and likely the cause of persistent left lower extremity weakness. During a psychiatric evaluation, the patient attributed his self-enucleation act to command auditory hallucinations and was transferred to an inpatient psychiatric unit for continuing care. At discharge, the patient was alert but confused with a GCS of 14. Motor strength was 4+/5 in the right hemibody, 4/5 in the left upper extremity, and 2/5 proximally, and 0/5 distally in the left lower extremity. The patient was given instructions to follow-up for orbital reconstruction with dermis fat graft.
Figure 3: Axial diffusion-weighted imaging image showing a hyperdensity in the right supplementary motor area, indicating an ischemic infarct

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Figure 4: Coronal digital subtraction angiography image with injection into the left internal carotid artery demonstrating filling of the right anterior cerebral and middle cerebral arteries through the anterior communicating artery

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Figure 5: Repeat axial computed tomography head image on posttrauma day 28 showing a hypodensity in the right supplementary motor area, consistent with a subacute infarct

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  Discussion Top


We present a unique case of self-enucleation, leading to both SAH and ICA dissection/thrombosis and subsequent coma (GCS 6), followed by a dramatic initial recovery with conservative management only. We also conducted a comprehensive review of the English-language literature for previous cases of self-enucleation that resulted in intracranial complications [Table 1].
Table 1: Summary of literature reporting cases of self-enucleation associated with intracranial complications

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We hypothesize that the ICA dissection associated with self-enucleation is a consequence of the traumatic OA avulsion at its take-off from the ICA.[8],[9] It leads to hemorrhage in the basilar cisterns mimicking a pattern seen in aneurysmal rupture. Subsequent ICA occlusion results from disruption of the arterial wall, facilitating an acute thrombotic event. Gauger et al. postulated that the dissection may result from direct force transmission to the ICA during enucleation.[4] ICA dissection with subsequent occlusion may constitute a life-threatening event in patients without adequate collateral blood flow.[22],[23]

Our patient suffered from an ischemic infarct in the right supplementary motor area, which was the likely cause of the left lower extremity weakness. Ischemic infarcts presenting within 2 weeks following SAH are concerning for vasospasm, as in Kotlus and Lo case,[5] which involved bilateral vasospastic strokes following self-enucleation. To assess for vasospasm in our patient, TCD measurements were performed daily and showed persistently elevated flow velocities in multiple arteries, but a subsequent DSA showed no evidence of vasospasm. While TCD is a convenient noninvasive test to screen for cerebral vasospasm,[24],[25] DSA remains the gold standard and has a higher diagnostic accuracy.[26] We believe that our patient's infarct was likely not due to vasospasm but rather due to an embolic event precipitated by ICA dissection and occlusion.[27]

A complication commonly reported in previous cases of self-nucleation but not seen in our patient is contralateral temporal field deficit (CTFC).[6],[12],[15],[21],[28] CTFC indicates chiasmal injury from avulsion of the optic nerve at its intracranial segment or more than 4 cm proximal to the optic globe and may improve with steroids.[28],[29]


  Conclusion Top


Self-enucleation is a dramatic and unfortunate complication of acute psychosis that presents as a neurologic, ophthalmologic, and psychiatric emergency. We reported a unique case of self-enucleation resulting in both SAH and ICA dissection and occlusion that improved initially with conservative management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patients have given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

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Castro HM, Alvarez J, Bota RG, Yonkers M, Tao J. A case of attempted bilateral self-enucleation in a patient with bipolar disorder. Ment Illn 2017;9:7141.  Back to cited text no. 3
    
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Gauger EH, Sobel RK, Allen RC. Complications and outcomes after autoenucleation. Curr Opin Ophthalmol 2015;26:429-38.  Back to cited text no. 4
    
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Kotlus BS, Lo MW. Subarachnoid hemorrhage and vasospastic stroke after self-enucleation. Ophthalmic Plast Reconstr Surg 2007;23:425-7.  Back to cited text no. 5
    
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Tuwir I, Chako E, Brosnahan D, Cassidy L. Drug induced autoenucleation with resultant chiasmal damage. Br J Ophthalmol 2005;89:121.  Back to cited text no. 6
    
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Dilly JS, Imes RK. Autoenucleation of a blind eye. J Neuroophthalmol 2001;21:30-1.  Back to cited text no. 12
    
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Eisenhauer GL. Self-inflicted ocular removal by two psychiatric inpatients. Hosp Community Psychiatry 1985;36:189-91.  Back to cited text no. 13
    
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Khan JA, Buescher L, Ide CH, Pettigrove B. Medical management of self-enucleation. Arch Ophthalmol 1985;103:386-9.  Back to cited text no. 14
    
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Krauss HR, Yee RD, Foos RY. Autoenucleation. Surv Ophthalmol 1984;29:179-87.  Back to cited text no. 15
    
16.
Leibovitch I, Pietris G, Casson R, Selva D. Oedipism: Bilateral self-enucleation. Am J Emerg Med 2006;24:127-8.  Back to cited text no. 16
    
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Murphy M, Nathan M, Lee E, Parsons B, Gunasekera L. Oedipism: Auto-enucleation in a schizophrenic patient. Ir J Psychol Med 2006;23:159-60.  Back to cited text no. 17
    
18.
Rosenthal JM, Carrasco J, Rittenhouse DW, Bilyk JR. Autoenucleation resulting in carotid thrombosis, subdural hemorrhage, stroke, and death. Orbit 2011;30:308-10.  Back to cited text no. 18
    
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Schargus M, Schneider E, Klink T. Autoenucleation in a 84-year-old dementia patient. Int Ophthalmol 2009;29:281-3.  Back to cited text no. 19
    
20.
Shah M, Sun L, Elmann S, Vrcek I, Mancini R, Kim HJ, et al. Self-inflicted enucleations: Clinical features of seven cases. Orbit 2017;36:154-8.  Back to cited text no. 20
    
21.
Tabatabaei S, Soleimani M, Khodabandeh A. A case of autoenucleation associated with a contralateral field defect. Orbit (Amsterdam, Netherlands) 2011;30:165-8.  Back to cited text no. 21
    
22.
Hedera P, Bujdakova J, Traubner P. Effect of collateral flow patterns on outcome of carotid occlusion. Eur Neurol 1995;35:212-6.  Back to cited text no. 22
    
23.
Sundaram S, Kannoth S, Thomas B, Sarma PS, Sylaja PN. Collateral assessment by CT angiography as a predictor of outcome in symptomatic cervical internal carotid artery occlusion. AJNR Am J Neuroradiol 2017;38:52-7.  Back to cited text no. 23
    
24.
Li DD, Chang JY, Zhou CX, Cui JB. Clinical diagnosis of cerebral vasospasm after subarachnoid hemorrhage by using transcranial Doppler sonography. Eur Rev Med Pharmacol Sci 2018;22:2029-35.  Back to cited text no. 24
    
25.
Mastantuono JM, Combescure C, Elia N, Tramer MR, Lysakowski C. Transcranial Doppler in the diagnosis of cerebral vasospasm: An updated meta-analysis. Crit Care Med 2018;46:1665-72.  Back to cited text no. 25
    
26.
Arias EJ, Vajapey S, Reynolds MR, Chicoine MR, Rich KM, Dacey RG, Jr., et al. Utility of screening for cerebral vasospasm using digital subtraction angiography. Stroke 2015;46:3137-41.  Back to cited text no. 26
    
27.
Chen CJ, Green IE, Worrall BB, Southerland AM. Cerebral collaterals and stroke in patients with isolated carotid artery dissections. J Clin Neurosci 2020;72:158-62.  Back to cited text no. 27
    
28.
Parmar B, Edmunds B, Plant G. Traumatic enucleation with chiasmal damage: Magnetic resonance image findings and response to steroids. Br J Ophthalmol 2002;86:1317-8.  Back to cited text no. 28
    
29.
Suzuki N, Fujitsu K, Tanaka N, Sekino T, Kuwabara T, Yuda K. Traumatic enucleation of the eye ball—report of a case and considerations concerning the pathogenic mechanism of intracranial complications. No Shinkei Geka 1988;16:1293-7.  Back to cited text no. 29
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1]



 

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