Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 13–15

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Interdisciplinary Neurosurgery:Advanced Techniques and Case Management

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Case Reports & Case Series (CRP)

Spontaneous subdural hematoma associated to Duret hemorrhage☆,☆☆

William Alves Martins, MD a,⁎, Alice Becker Teixeira, MD a, Thomas More Frigeri, MD b,Eliseu Paglioli, MD, PhD b

a Department of Neurology, Hospital São Lucas, Pontifícia Universidade Católica do Rio Grande do Sul (PUCRS), Porto Alegre, RS, Brazilb Department of Neurosurgery, Hospital São Lucas, Pontifícia Universidade Católica do Rio Grande do Sul (PUCRS), Porto Alegre, RS, Brazil

a b s t r a c ta r t i c l e i n f o

☆ This article is in accord to all coauthors and its manin any other medical journal.☆☆ Conflict of interest: All authors declare to have no

⁎ Corresponding author at: Department of NeurologyAvenida Ipiranga 6690, 90610–000, Porto Alegre, RS,2760.

E-mail address: walvesm.br@gmail.com (W.A. Mar

http://dx.doi.org/10.1016/j.inat.2014.11.0032214-7519/© 2014 The Authors. Published by Elsevier

Article history:Received 26 August 2014Revised 11 November 2014Accepted 16 November 2014

Keywords:Acute spontaneous subdural hematomaCortical artery ruptureDuret hemorrhage

Subdural hematoma (SH) is a neurosurgical emergency, usually caused by head trauma. Non-traumatic causesinclude aneurysm or arterial–venous malformation rupture, coagulopathy and others. We report the case of a66 year-old man who developed apparently unprovoked signs of increased intracranial pressure. Braincomputed tomography scan showed an acute spontaneous SH, surgically treated. Throughout surgery, aruptured cortical artery with intensive bleeding appeared andwas cauterized. After surgery, patient remainedcomatose and a new CT demonstrated Duret hemorrhage at the brainstem. Acute spontaneous SH of arterialorigin is rare and highly lethal, in which a good prognosis relies on early diagnosis and treatment.© 2014 The Authors. Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license

(http://creativecommons.org/licenses/by-nc-nd/3.0/).

Introduction

Acute subdural hematoma (SH) is a common neurosurgicalemergency, usually due to rupture of superficial cerebral or corticalbridging veins during severe head trauma. Non-traumatic causesinclude aneurysm or arterial–venous malformation (AVM) rupture,coagulopathy, cocaine abuse, metastatic cancer, Moya-Moya diseaseand others [1–4].

Acute spontaneous SH of arterial origin is a rare disorder, causedby disruption of a perisylvian artery without history of head traumaand evidence of AVM or aneurysm [1,2]. Evolution is sudden anddramatic neurological deterioration may be seen within minutes.There is no clear precipitant, but minor head trauma is thought to playa role in rupturing an adhered cortical artery, leading to massivesubdural bleeding [3,4]. Prompt neurosurgical evacuation is crucial toa good outcome [5,6]. We report a case spontaneous SH of arterialorigin associated to Duret hemorrhage after sudden rise in intracra-nial pressure and briefly review the current literature.

Case report

A 66-year-old male had been admitted at Hospital São Lucas forcomplementary investigation of a cholestatic syndrome due to

uscript has not been submitted

conflict of interest., Hospital São Lucas da PUCRS,Brazil. Tel./fax: +55 51 3320

tins).

B.V. This is an open access article u

pancreatic cancer. On the 15th day of admission, after coughing, hesuddenly developed severe headache, vomiting, right-side weaknessand rapidly evolved with altered mental status. At the time, systemicinvestigation with CT scans of thorax, abdominal and pelvic regions,along with bone scintigraphy, had been negative for metastaticinvolvement. No history of head trauma was evident and he did notdisplay any prior neurological signs suggestive of metastatic disease.General inspection showed that he was jaundiced and mildlyundernourished. Neurological examination showed anisocoria witha dilated (5 mm) left pupil, unresponsive to light. Glasgow coma scale(GCS) was 5/15 and he presented clear right-side weakness. A mildexternal deviation of the left eye could be seen, suggestive of IIIrdnerve palsy. After orotraqueal intubation, brain CT scan showed noevidence of metastatic disease, but uncovered an acute SH in the leftfrontoparietal region with signs of active bleeding, causing significantmidline shift and transtentorial herniation (Fig. A, B).

Laboratory tests were normal, except for mild normochromicanemia and conjugated hyperbilirubinemia. Coagulation studies didnot show any abnormalities and he was only on prophylactic heparin,without any history of hemorrhagic diatheses. Platelet count was318,000/μl, within normal range (150,000–400,000).

Surgery was immediately undertaken, using a left pterionalcraniotomy to access the hematoma. After opening the Dura mater,we encountered continuous arterial bleeding from a rupturedsuperficial cortical artery (Video). There was no sign of AVM oraneurism at the site and this arterywas easily cauterized, immediatelystopping the bleeding. After evacuation of the extensive SH, thepatient was admitted to intensive care unit.

One day latter, subsequent CT scan showed almost completeresolution of SH, but displayed mild hemorrhage in the leftparahippocampus and midbrain (Fig. 1C, D). Patient remained

nder the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/3.0/).

Fig. Brain CT scan. A) Extensive SH in left convexity and mass effect before surgery; B) Transtentorial herniation (red arrow) and subdural hematoma in the temporal lobe (asterix);C and D) Post-operative CT scan showing hemorrhagic foci in left parahippocampal gyrus and midbrain (white arrows).

14 W.A. Martins et al. / Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 13–15

comatose and died nine days after surgical intervention, due tourinary tract septicemia.

Discussion

Acute spontaneous SH of arterial origin had been first described byMunro in 1934, but it was only seen as a unique syndrome by Tallalaet al. in 1971 [2–4]. In 1988, Tokoro and colleagues proposed fourdiagnostic criteria: (a) no history of head trauma, (b) no cerebralcortex damage, (c) no evidence of ruptured aneurysm or AVM, and(d) identification of arterial hemorrhage during operation [1].Following these criteria, about 91 cases have been reported inliterature until now [6]. Our case satisfies Tokoro’s criteria for thediagnosis of acute spontaneous SH of arterial origin and highlights thesudden, acute and dramatic onset, probably induced by an increase inintracranial pressure following Valsalva-maneuver.

The pathophysiological mechanism behind this uncommon con-dition is controversial, but could involve: a) avulsion of a small arterybranching off in a right angle; b) rupture of a corticodural bridgingartery or c) adhesions between a cortical artery and the Dura mater[2–4]. Rather than lack of trauma, some authors state that to rupture

these arteries would actually take minimum trauma [2]. Our patient’scough could have generated a small increase in intracranial pressure,just enough to displace brain tissue within the cranium and disrupt analready adhered artery.

Acute spontaneous SH usually manifests as sudden unexplainedcoma, severe headache, vomiting, focal neurological deficits andseizure [1–4], mimicking stroke or subarachnoid hemorrhage [3].Surgical evaluation should be urgent and a brain CT scan be performedas soon as possible after clinical stabilization, to guide furthertherapeutic measures.

Mortality rate ranges from 25% to 90% in reported series and maybe associated with a lower GCS at presentation and delay in surgicaltreatment [3,6]. In a recent review, Coombs and colleagues found 91cases of acute spontaneous SH of arterial origin reported on literature,corresponding for 61.5% of all acute spontaneous SHs with an overallmortality rate of 36.7% [6]. The mean age was 61 years old and it wastwice more frequent in males [2]. Known risk factors include age over50 years, alcohol abuse and systemic arterial hypertension [2–6].Spontaneous and traumatic SHs actually share these risk factors. Someauthors argue that patients with coagulopathy [3,4] should notreceive the diagnosis of spontaneous SH, while others say thatcoagulopathy is only a contributor to a worse prognosis [2]. Our

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patient had a cholestatic syndrome, however he did not show anysigns of coagulopathy or thrombocytopenia. The only anticoagulantdrug in use was prophylactic non-fractionated heparin 5000 U twice aday, which probably did not play a role in his presentation.

Sudden development of SH with continuous intrahematomatousbleeding predisposes to rapid temporal lobe herniation and mayexplain the post-surgical appearance of Duret hemorrhage (DH) inour patient. Duret’s phenomenon is actually a secondary hemorrhagicinfarction of midbrain [7], resulting from compression and shearing ofbrainstem penetrating arteries by herniated parahippocampal gyrus[8]. Risk factors for development of DH include fast raising inintracranial pressure, old age, volume of the lesion and abruptchanges in intracranial pressure, such as produced by lumbarpuncture or decompressive craniotomy [8,9]. The latter would causesudden restoration of blood flow and reperfusion injury to ischemicbrainstem. Nedergaard and colleagues studied the frequency of DT inacute vascular lesions [9]. In their study, DH developed in 45% inspontaneous cerebral hemorrhage, 36% in ruptured aneurysm, and15% in ischemic stroke [9]. As several important axonal pathways aredamaged in DH, it carries a poor prognosis [7], although some reportonly mild deficits, such as internuclear ophthalmoplegia [10].Combination of acute SH of arterial origin and DH is extremelyuncommon, despite its quick evolution to transtentorial herniationand has been reported only once [10].

Differential diagnosis in our patient comprises dural metastasis,cerebral venous thrombosis (CVT) and AVM or aneurism as theunderlying etiology of SH. Dural metastasis causing or simulating SHis rare [11], mainly seen in patients with advancedmalignancy, unlikeour patient, who had limited neoplastic disease. Other possiblecancer-related complication is CVT [12]. Studies show that 11% of allCVTs might be related to malignancy [13]. Nevertheless, CVT has onlyrarely been associated to SH [14], both of which are usually caused byintracranial hypotension, not fit for our patient presentation.Likewise, isolated SH is a rare presentation of AVM or aneurism[15,16]. Though our case did not allow assessment of vascularmalformation (e.g. MRI and MRA), given the clinical acuity, we feelthat visualization of a distal ruptured cortical artery during surgerywithout morphological anomalies or AVM, supports the diagnosis ofacute spontaneous SH of arterial origin.We do not feel that the arterialbleeding was artificial as it was noted instantaneously as an arterial"pumper" through the subdural clot after gentle opening andelevation of the dura mater, as demonstrated in the surgical video.

Acute spontaneous SH of arterial origin is a rare and lethaldisorder. Our case highlights the dramatic evolution of this condition,showing that immediate diagnostic approach and intervention areessential to give these patients appropriate care, since early surgicaldrainage is crucial to a good outcome.

Supplementary data to this article can be found online at http://dx.doi.org/10.1016/j.inat.2014.11.003.

References

[1] Tokoro K, Nakajima F, Yamataki A. Acute subdural hematoma of arterial origin.Surg Neurol 1988;29:159–63.

[2] Depreitere B, Van Calenbergh F, Van Loon J. A clinical comparison of non-traumaticacute subdural hematomas either related to coagulopathy or of arterial originwithout coagulopathy. Acta Neurochir (Wien) 2003;145:541–6.

[3] Chhiber SS, Singh JP. Acute spontaneous subdural hematoma of arterial origin: areport of four cases and review of literature. Neurol India 2010;58:654–8.

[4] Akioka N, Fukuda O, Takaba M, Kameda H, Saito T, Endo S. Clinical investigation ofacute spontaneous subdural hematoma cases. J Stroke Cerebrovasc Dis 2007;16(3):109–13.

[5] Gelabert-González M, Fernández-Villa JM, Iglesias-Pais M, et al. Acute spontaneoussubdural haematoma of arterial origin. Neurocírugía 2004;15:165–70.

[6] Coombs JB, Coombs BL, Chin EJ. Acute spontaneous subdural hematoma in amiddle-aged adult: case report and review of the literature. J Emerg Med 2014:1–5.

[7] Moore SA, Mittal MK, Rabinstein AA. Duret hemorrhage following subduralhematoma evacuation. JAMA Neurol 2013;70(4):518.

[8] Chew KL, Baber Y, Iles L, O'Donnell C. Duret hemorrhage: demonstration ofruptured paramedian pontine branches of the basilar artery on minimallyinvasive, whole body postmortem CT angiography. Forensic Sci Med Pathol2012;8(4):436–40.

[9] Nedergaard M, Klinklen L, Paulson O. Secondary brain stem hemorrhage in stroke.Stroke 1983;14(4):501–5.

[10] Rouhl RPW, Postma AA. Teaching neuroimages: Duret hemorrhage due tobilateral subdural hematomas causing internuclear ophthalmoplegia. Neurology2012;79:e101.

[11] Sato M, Saito T, Yamaguchi K, Sakuma H. A case of acute subdural hematoma dueto dural metastasis from malignant pleural mesothelioma. No Shinkei Geka 1994;22(3):247–51.

[12] Raizer JJ, DeAngelis LM. Cerebral sinus thrombosis diagnosed by MRI and MRvenography in cancer patients. Neurology 2000;54(6):1222–6.

[13] Korathanakhun P, Sathirapanya P, Geater SL, Petpichetchian W. Predictors ofhospital outcome in patients with cerebral venous thrombosis. J StrokeCerebrovasc Dis 2014;S1052–3057(14):00302–4.

[14] Akins PT, Axelrod YK, Ji C, Ciporen JN, Arshad ST, Hawk MW, et al. Cerebral venoussinus thrombosis complicated by subdural hematomas: case series and literaturereview. Surg Neurol Int 2013;19(4):85.

[15] Ogawa K, Oishi M, Mizutani T, Maejima S, Mori T. Dural arteriovenous fistula onthe convexity presenting with pure acute subdural hematoma. Acta Neurol Belg2010;110(2):190–2.

[16] Mansour O, Hassen T, Fathy S. Acute aneurismal bilateral subdural haematomawithout subarachnoid haemorrhage: a case report and review of the literature.Case Rep Neurol Med 2014;2014:260853.