AbstractSubdural hematoma (SH) is a neurosurgical emergency, usually caused by head trauma. Non-traumatic causes include aneurysm or arterial–venous malformation rupture, coagulopathy and others. We report the case of a 66year-old man who developed apparently unprovoked signs of increased intracranial pressure. Brain computed tomography scan showed an acute spontaneous SH, surgically treated. Throughout surgery, a ruptured cortical artery with intensive bleeding appeared and was cauterized. After surgery, patient remained comatose and a new CT demonstrated Duret hemorrhage at the brainstem. Acute spontaneous SH of arterial origin is rare and highly lethal, in which a good prognosis relies on early diagnosis and treatment

Frigeri, Thomas More

Martins, William Alves

Paglioli, Eliseu

Teixeira, Alice Becker

Elsevier - Publisher Connector 

Spontaneous subdural hematoma associated to Duret hemorrhage 

Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 13–15
Contents lists available at ScienceDirect
Interdisciplinary Neurosurgery:
Advanced Techniques and Case Management
j ourna l homepage: www. inat - journa l .comCase 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, Brazil
b 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 man
in any other medical journal.
☆☆ Conﬂict of interest: All authors declare to have no
⁎ Corresponding author at: Department of Neurology
Avenida 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.2014.11.003
2214-7519/© 2014 The Authors. Published by ElsevierArticle history:
Received 26 August 2014
Revised 11 November 2014
Accepted 16 November 2014
Keywords:
Acute spontaneous subdural hematoma
Cortical artery rupture
Duret hemorrhageSubdural hematoma (SH) is a neurosurgical emergency, usually caused by head trauma. Non-traumatic causes
include aneurysm or arterial–venous malformation rupture, coagulopathy and others. We report the case of a
66 year-old man who developed apparently unprovoked signs of increased intracranial pressure. Brain
computed tomography scan showed an acute spontaneous SH, surgically treated. Throughout surgery, a
ruptured cortical artery with intensive bleeding appeared andwas cauterized. After surgery, patient remained
comatose and a new CT demonstrated Duret hemorrhage at the brainstem. Acute spontaneous SH of arterial
origin 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 neurosurgical
emergency, usually due to rupture of superﬁcial cerebral or cortical
bridging veins during severe head trauma. Non-traumatic causes
include aneurysm or arterial–venous malformation (AVM) rupture,
coagulopathy, cocaine abuse, metastatic cancer, Moya-Moya disease
and others [1–4].
Acute spontaneous SH of arterial origin is a rare disorder, caused
by disruption of a perisylvian artery without history of head trauma
and evidence of AVM or aneurysm [1,2]. Evolution is sudden and
dramatic neurological deterioration may be seen within minutes.
There is no clear precipitant, but minor head trauma is thought to play
a role in rupturing an adhered cortical artery, leading to massive
subdural bleeding [3,4]. Prompt neurosurgical evacuation is crucial to
a good outcome [5,6]. We report a case spontaneous SH of arterial
origin 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 for
complementary investigation of a cholestatic syndrome due touscript 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 upancreatic cancer. On the 15th day of admission, after coughing, he
suddenly developed severe headache, vomiting, right-side weakness
and rapidly evolved with altered mental status. At the time, systemic
investigation with CT scans of thorax, abdominal and pelvic regions,
along with bone scintigraphy, had been negative for metastatic
involvement. No history of head trauma was evident and he did not
display any prior neurological signs suggestive of metastatic disease.
General inspection showed that he was jaundiced and mildly
undernourished. Neurological examination showed anisocoria with
a dilated (5 mm) left pupil, unresponsive to light. Glasgow coma scale
(GCS) was 5/15 and he presented clear right-side weakness. A mild
external deviation of the left eye could be seen, suggestive of IIIrd
nerve palsy. After orotraqueal intubation, brain CT scan showed no
evidence of metastatic disease, but uncovered an acute SH in the left
frontoparietal region with signs of active bleeding, causing signiﬁcant
midline shift and transtentorial herniation (Fig. A, B).
Laboratory tests were normal, except for mild normochromic
anemia and conjugated hyperbilirubinemia. Coagulation studies did
not show any abnormalities and he was only on prophylactic heparin,
without any history of hemorrhagic diatheses. Platelet count was
318,000/μl, within normal range (150,000–400,000).
Surgery was immediately undertaken, using a left pterional
craniotomy to access the hematoma. After opening the Dura mater,
we encountered continuous arterial bleeding from a ruptured
superﬁcial cortical artery (Video). There was no sign of AVM or
aneurism at the site and this arterywas easily cauterized, immediately
stopping the bleeding. After evacuation of the extensive SH, the
patient was admitted to intensive care unit.
One day latter, subsequent CT scan showed almost complete
resolution of SH, but displayed mild hemorrhage in the left
parahippocampus and midbrain (Fig. 1C, D). Patient remainednder 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) 13–15comatose and died nine days after surgical intervention, due to
urinary tract septicemia.Discussion
Acute spontaneous SH of arterial origin had been ﬁrst described by
Munro in 1934, but it was only seen as a unique syndrome by Tallala
et al. in 1971 [2–4]. In 1988, Tokoro and colleagues proposed four
diagnostic criteria: (a) no history of head trauma, (b) no cerebral
cortex 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 in
literature until now [6]. Our case satisﬁes Tokoro’s criteria for the
diagnosis of acute spontaneous SH of arterial origin and highlights the
sudden, acute and dramatic onset, probably induced by an increase in
intracranial pressure following Valsalva-maneuver.
The pathophysiological mechanism behind this uncommon con-
dition is controversial, but could involve: a) avulsion of a small artery
branching off in a right angle; b) rupture of a corticodural bridging
artery or c) adhesions between a cortical artery and the Dura mater
[2–4]. Rather than lack of trauma, some authors state that to rupturethese arteries would actually take minimum trauma [2]. Our patient’s
cough could have generated a small increase in intracranial pressure,
just enough to displace brain tissue within the cranium and disrupt an
already adhered artery.
Acute spontaneous SH usually manifests as sudden unexplained
coma, severe headache, vomiting, focal neurological deﬁcits and
seizure [1–4], mimicking stroke or subarachnoid hemorrhage [3].
Surgical evaluation should be urgent and a brain CT scan be performed
as soon as possible after clinical stabilization, to guide further
therapeutic measures.
Mortality rate ranges from 25% to 90% in reported series and may
be associated with a lower GCS at presentation and delay in surgical
treatment [3,6]. In a recent review, Coombs and colleagues found 91
cases of acute spontaneous SH of arterial origin reported on literature,
corresponding for 61.5% of all acute spontaneous SHs with an overall
mortality rate of 36.7% [6]. The mean age was 61 years old and it was
twice more frequent in males [2]. Known risk factors include age over
50 years, alcohol abuse and systemic arterial hypertension [2–6].
Spontaneous and traumatic SHs actually share these risk factors. Some
authors argue that patients with coagulopathy [3,4] should not
receive the diagnosis of spontaneous SH, while others say that
coagulopathy is only a contributor to a worse prognosis [2]. Our
15W.A. Martins et al. / Interdisciplinary Neurosurgery: Advanced Techniques and Case Management 2 (2015) 13–15patient had a cholestatic syndrome, however he did not show any
signs of coagulopathy or thrombocytopenia. The only anticoagulant
drug in use was prophylactic non-fractionated heparin 5000 U twice a
day, which probably did not play a role in his presentation.
Sudden development of SH with continuous intrahematomatous
bleeding predisposes to rapid temporal lobe herniation and may
explain the post-surgical appearance of Duret hemorrhage (DH) in
our patient. Duret’s phenomenon is actually a secondary hemorrhagic
infarction of midbrain [7], resulting from compression and shearing of
brainstem penetrating arteries by herniated parahippocampal gyrus
[8]. Risk factors for development of DH include fast raising in
intracranial pressure, old age, volume of the lesion and abrupt
changes in intracranial pressure, such as produced by lumbar
puncture or decompressive craniotomy [8,9]. The latter would cause
sudden restoration of blood ﬂow and reperfusion injury to ischemic
brainstem. Nedergaard and colleagues studied the frequency of DT in
acute vascular lesions [9]. In their study, DH developed in 45% in
spontaneous cerebral hemorrhage, 36% in ruptured aneurysm, and
15% in ischemic stroke [9]. As several important axonal pathways are
damaged in DH, it carries a poor prognosis [7], although some report
only mild deﬁcits, such as internuclear ophthalmoplegia [10].
Combination of acute SH of arterial origin and DH is extremely
uncommon, despite its quick evolution to transtentorial herniation
and has been reported only once [10].
Differential diagnosis in our patient comprises dural metastasis,
cerebral venous thrombosis (CVT) and AVM or aneurism as the
underlying etiology of SH. Dural metastasis causing or simulating SH
is rare [11], mainly seen in patients with advancedmalignancy, unlike
our patient, who had limited neoplastic disease. Other possible
cancer-related complication is CVT [12]. Studies show that 11% of all
CVTs might be related to malignancy [13]. Nevertheless, CVT has only
rarely been associated to SH [14], both of which are usually caused by
intracranial 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 vascular
malformation (e.g. MRI and MRA), given the clinical acuity, we feel
that visualization of a distal ruptured cortical artery during surgery
without morphological anomalies or AVM, supports the diagnosis of
acute spontaneous SH of arterial origin.We do not feel that the arterial
bleeding was artiﬁcial as it was noted instantaneously as an arterial
"pumper" through the subdural clot after gentle opening and
elevation of the dura mater, as demonstrated in the surgical video.Acute spontaneous SH of arterial origin is a rare and lethal
disorder. Our case highlights the dramatic evolution of this condition,
showing that immediate diagnostic approach and intervention are
essential to give these patients appropriate care, since early surgical
drainage 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.
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Spontaneous subdural hematoma associated to Duret hemorrhage

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