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Clinical Case Report Medicine ®

Cerebral venous thrombosis presented with


symmetrical crescent-shaped intracranial
hemorrhage in alcoholic liver disease
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Case reports
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Lingjia Xu, MMa, Guoping Fu, MMa,*

Abstract
Rationale: Cerebral venous thrombosis (CVT) is a relatively uncommon but fatal disease. It can be caused by a variety of
hereditary or acquired thrombotic diseases. Initial presentation with intracranial hemorrhage (ICH) in CVT is rare but can further
complicate the therapeutic measures and prognosis. Cases of CVT presented with ICH in patients with alcoholic liver disease
(ALD) have not been described in the literature, and it might be related with hemostatic abnormalities in ALD patients.
Patient concerns: We report 2 cases of men admitted to our hospital who were diagnosed with CVT but initially presented
with symmetrical crescent-shaped ICH; both of them were ALD patients.
Diagnoses: Cerebral imaging revealed extended CVT in both cases. The first case was a 64-year-old man with ALD deteriorated
with unconsciousness and convulsions; computed tomography showed symmetrical crescent-shaped ICH in the right temporal
lobe, and magnetic resonance venography revealed CVT. Another 50-year-old man with ALD complained about dizziness and
weakness of his right limbs; computed tomography revealed symmetrical crescent-shaped ICH in bilateral parietal and occipital
lobes, and magnetic resonance venography revealed CVT.
Interventions: The first patient was referred to the endovascular thrombectomy. Both of them were treated with anticoagulation
treatment.
Outcomes: Favorable outcomes were observed in both patients.
Lessons: Symmetrical or multiple crescent-shaped ICH requires a high suspicion in the diagnosis of CVT; even with hemorrhage,
it is still important to initiate anticoagulation therapy promptly. The crescent-shaped ICH might be a new sign for CVT, and further
studies are needed in the underlying mechanisms of ALD and potential thrombophilia.
Abbreviations: ALD = alcoholic liver disease, CT = computed tomography, CVT = cerebral venous thrombosis, ICH = intracranial
hemorrhage, MRV = magnetic resonance venography, SSS = superior sagittal sinus.
Keywords: alcoholic liver disease, case reports, cerebral venous thrombosis, intracranial hemorrhage, magnetic resonance
venography

1. Introduction Initial presentation with intracerebral hemorrhage (ICH) in


CVT patients is rare but can further complicate the treatment
Cerebral venous thrombosis (CVT) is a rare but serious cerebro- and prognosis.[3]
vascular disease with high lethiferous and disable ratio.[1] It is Alcoholic liver disease (ALD) is one of the most prevalent
more common in young people, especially females. The etiology chronic liver diseases and the leading cause of cirrhosis after
of CVT is complex; it can be caused by a variety of medical viral hepatitis or nonalcoholic fatty liver disease; ALD is typ-
conditions and the majors are genetic or acquired coagulation ically diagnosed by a patient’s medical history, physical exam-
dysfunction diseases, such as infection, tumor, brain trauma, ination, imaging, laboratory tests, and liver biopsy is the gold
pregnancy, oral contraceptives, thrombophilia, and so on.[2] standard for diagnosing ALD.[4] Patients suffering from liver

The study was supported by grants from the Health Commission of Zhejiang * Correspondence: Guoping Fu, Department of Neurology, Shaoxing Second
Province (2024KY492). Hospital, The Second Affiliated Hospital of Shaoxing University Medical College,
The authors have no conflicts of interest to disclose. Shaoxing 312000, Zhejiang, China (e-mail: sxeysjnk@163.com).

All data generated or analyzed during this study are included in this published Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.
article [and its supplementary information files]. This is an open access article distributed under the Creative Commons
Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and
Written informed consents were obtained from the patients for the publication of reproduction in any medium, provided the original work is properly cited.
any potentially identifiable images or data included in this article.
How to cite this article: Xu L, Fu G. Cerebral venous thrombosis presented with
Supplemental Digital Content is available for this article. symmetrical crescent-shaped intracranial hemorrhage in alcoholic liver disease:
a
Department of Neurology, Shaoxing Second Hospital, The Second Case reports. Medicine 2024;103:10(e37441).
Affiliated Hospital of Shaoxing University Medical College, Shaoxing, Received: 29 November 2023 / Accepted: 9 February 2024
Zhejiang, China.
http://dx.doi.org/10.1097/MD.0000000000037441

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Xu and Fu • Medicine (2024) 103:10Medicine

illness experience decreased absorption of vitamin K, which 102 mm Hg, respectively. The real-time head CT found no obvi-
leads to the synthesis and depletion of various coagulation com- ous abnormalities. On laboratory examination, the D-dimer
ponents, ultimately causing an imbalance between coagulation concentration increased (619 ng/mL, normal < 500 ng/mL),
and anticoagulation function.[5] and the result of liver function test reported elevated alanine
Here we report 2 patients with CVT presented with sym- aminotransferase (120 U/L, normal < 40 U/L), aspartate ami-
metrical crescent-shaped ICH, and both have the underlying notransferase (66 U/L, normal < 35 U/L), and r-glutamyl trans-
diseases of ALD, whether the thrombophilia mediated by ALD ferase (144 U/L, normal < 45 U/L). Other blood tests including
leads to CVT, is the focus of discussion and future research blood routine test, thyroid and kidney function, rheumatism
direction. Timely computed tomography (CT) or magnetic reso- index, and tumor-related markers were all within the normal
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nance venogram (MRV) examination has a pivotal position for range. Urapidil hydrochloride injection and pantoprazole were
better diagnosis and clinical treatment to improve the prognosis applied emergently. Ten hours after admission, the patient sud-
of patients. denly lost consciousness with limb convulsions for about 5
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minutes. He became delirious, failed to respond when calling


and to cooperate in examination of muscle strength, and bilat-
2. Case reports eral Babinski sign was negative. Urgent reexamination of the
head CT showed symmetrical crescent-shaped ICH in the right
2.1. Case 1 temporal lobe (Fig. 1A). Since the hematoma shape was not in
A 64-year-old man who had a history of ALD (used to drink line with the general arterial hemorrhage, magnetic resonance
White wine 200 mL per day for about 40 years, equivalent to imaging and MRV were performed and then revealed throm-
166.4 g of pure alcohol per day), cirrhosis, and hypertension but bosis of the superior sagittal sinus (SSS), right transverse sinus,
had not received regular treatment, no family history of genetic and sigmoid sinus with corresponding hemorrhagic cerebral
diseases or traumatic history, went to our hospital’s emergency infarction (Fig. 1B–E). Emergent interventional thrombectomy
department for treatment after experiencing dizziness, nausea, was administrated. The patient was transferred to the inten-
vomiting, tinnitus, and drowsiness for 5 days without a fever sive care unit after the surgery with continuous anticoagulation
or convulsions. The physical examination found the patient therapy and 1 week later, he was transferred to a rehabilitation
was conscious, well-oriented, had normal pulse with regular institution with a modified Rankin Scale score of 3. He recov-
rhythm, and normal muscle strength, but the patient’s sys- ered well after the surgery with no significant sequela moni-
tolic and diastolic blood pressure measurements were 225 and tored in 2-month follow-up, his CT scan revealed hemorrhage

Figure 1. Case 1. (A and B) Non-contrast brain computed tomography (CT) scan and T1 hyperintensity of magnetic resonance imaging (MRI) suggest sym-
metrical crescent-shaped intracranial hemorrhage in the right temporal and occipital lobes (White box). (C) Fluid-attenuated inversion recovery (FLAIR) imaging
shows juxtacortical edema surrounding the hemorrhage. (D) Diffusion weighted imaging (DWI) shows massive cerebral infarction throughout the right temporal
and occipital lobes. (E) Magnetic resonance venography (MRV) imaging shows cerebral venous sinus thrombosis of the superior sagittal sinus, right transverse
sinus, and sigmoid sinus (arrows). (F) CT examination obtained at 2-month follow-up.

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absorption and an ancient infarction in the right temporal lobe CVT impedes venous blood drainage, leading to increased venous
(Fig. 1F and Figure S1, Supplemental Digital Content, http:// pressure and subsequent brain tissue edema[7]; simultaneously,
links.lww.com/MD/L845 which indicates the timeline). metabolites build up and interfere with neurovascular oxygen-
ation, destroying the walls of capillaries and veins; this compro-
mises the blood-brain barrier, causing blood to leak through and
2.2. Case 2 causing hemorrhagic transformation of cerebral venous infarc-
tion[8]; on the other hand, due to the absence of valve in cortical
A 50-year-old man who was also an alcoholic (yellow rice
veins and smooth muscle in tunica media, the increased venous
wine ingestion for over 30 years, with a daily consumption of
pressure during CVT leads to gradual dilation and even rupture
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1000 ml, equivalent to 128 g of pure alcohol per day) and diag-
of these veins, making blood extravasation to the brain paren-
nosed ALD presented to our emergency room with a sudden
chyma and eventually hemorrhage in multiple brain lobes.[9]
onset of dizziness accompanied by blurred vision, gradually pro-
After viral hepatitis and nonalcoholic fatty liver disease, ALD
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gressive right limbs fatigue after getting up in the morning. He


is a kind of chronic liver disease and the primary cause of cirrho-
had no other diseases and drug use history, no family history, no
sis.[10] A further potential cause of thromboembolism is liver dys-
genetic diseases, or traumatic history. During admission phys-
function; studies have reported that various types of liver diseases
ical examination reported the patient was conscious, and had
are associated with an increased risk of developing thrombosis[11];
normal body temperature, respiratory rate, blood pressure, but
nevertheless, the mechanisms underlying how liver dysfunction
right limbs hemiplegia (grade 1 of muscle strength), and positive
leads to hypercoagulable states and how they translate into
right Babinski sign. His electrocardiography showed a pulse of
thrombotic events remain to be fully understood. In the issue of
47 beats per minute indicating sinus bradycardia. The result of
ALD, in addition to potentiating the growth of Gram-negative
testing for the coagulation profile solely revealed a high con-
bacteria in the intestine and increasing intestinal permeability,
centration of D-dimer (1846 ng/mL). Residual blood test exclu-
alcohol consumption can damage liver cells by encouraging the
sively showed abnormal liver function with elevated alanine
accumulation of acetaldehyde and other reactive oxygen species
aminotransferase, aspartate aminotransferase, and r-glutamyl
in the liver, which may cause oxidative stress, metabolism disor-
transferase (156, 78, and 256 U/L). Others including blood
ders, and liver cell death. This ultimately raises the level of endo-
routine test, thyroid and kidney function, rheumatism index,
toxin in peripheral blood and causes inflammation, necrosis, and
and tumor-related markers reported no obvious abnormalities.
fibrosis in the liver.[10,12] Proteases involved in the fibrinolytic sys-
His abdominal CT showed cirrhosis with liver lobar imbalance
tem, anticoagulation factors, and coagulation factors are all syn-
and widened portal vein (Fig. 2). Brain CT showed bilateral
thesized in large quantities in the liver. Due to variable degrees of
parietal and occipital lobes hemorrhage in consistent with the
injury or disruption, liver cells’ capacity to produce coagulation
symmetrical crescent-shape (Fig. 3A and B). The brain magnetic
and anticoagulation factors declines in ALD, leading to abnor-
resonance imaging found corresponding multifocal cerebral
malities of coagulation and anticoagulation mechanisms. In this
infarction with hemorrhagic transformation, MRV found SSS
context, patients with severe ALD may have both a higher risk of
thrombosis (Fig. 3C–E). Anticoagulant therapy with daltepa-
developing hemorrhage and thrombosis.[13]
rin sodium, dehydration cranial pressure therapies with glyc-
CVT with concomitant ALD has not been reported yet; in the
erol fructose and mannitol were applied, together with gastric
present report, both patients had ALD and a high D-dimer con-
mucosal protective agent, intravenous fluids, and rehabilitation
centration without other coagulation indicators of abnormali-
at bedside. The patient’s condition improved after anticoagula-
ties were detected. Whether the CVT represents a major threat
tion treatment in 1-month follow-up, and the CT showed hem-
of hemostatic abnormalities among ALD patients requires fur-
orrhage absorption (Fig. 3F and Figure S2, Supplemental Digital
ther exploration with more cases. Common cerebral hemato-
Content, http://links.lww.com/MD/L846 which indicates the
mas are often single, round, and oval-shaped, in line with the
timeline).
distribution of cerebral arterial blood vessels, while ICH with
diverse forms like the shape of cashew nuts, carob is often
detected in CVT with hemorrhagic transformation.[7,14] In our
3. Discussion
cases, the crescent-shaped juxtacortical hemorrhages may be
CVT is a relatively uncommon but fatal disease. Therefore, early related to the orientation of the subcortical vein along the arcu-
diagnosis and prompt management are critical, and knowledge ate fiber. The superficial and deep systems of the brain’s venous
of its radiological features and potential pathology are essential drainage merge into the subcortical veins and SSS in the cor-
to help it. ICH occurs in 30% to 40% of patients with CVT; tex,[15] and when the SSS is blocked by thrombus, the pressure
CVT initially presented with ICH predicts a poorer prognosis and of the subcortical veins increases and it is easy to rupture and
might be the leading cause of death in CVT patients,[6] however, it lead to juxtacortical hemorrhages. Clinically, the symmetrical
remains elusive about the specific pathophysiology. On one hand, crescent-shaped ICH in the aforementioned images, whether

Figure 2. Abdominal CT revealed cirrhosis, liver lobar imbalance, and widened portal vein in Case 2. CT = computed tomography.

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Figure 3. Case 2. (A and B) The brain CT scan shows symmetrical crescent-shaped intracranial hemorrhage in bilateral parietal and occipital lobes (White
boxes). (C and D) DWI and FLAIR show corresponding multifocal cerebral infarction with hemorrhagic transformation. (E) MRV suggests superior sagittal sinus
thrombosis (arrow). (F) CT examination obtained at 1-month follow-up. CT = computed tomography, DWI = diffusion weighted imaging, FLAIR = fluid-attenuated
inversion recovery, MRV = magnetic resonance venography.

or not it is in the same hemisphere, requires a high degree of [2] Sun J, He Z, Nan G. Cerebral venous sinus thrombosis presenting with
suspicion when diagnosing CVT, and it is highly recommended multifocal intracerebral hemorrhage and subarachnoid hemorrhage: a
to perform MRV examination to confirm the diagnosis earlier case report. Medicine (Baltim). 2018;97:e13476.
[3] Kobal J, Cankar K, Ivanusic K, et al. Early isolated subarachnoid hem-
and improve the prognosis. Moreover, additional research is
orrhage versus hemorrhagic infarction in cerebral venous thrombosis.
required to address the possibility of thrombophilia and ALD. Radiol Oncol. 2022;56:303–10.
[4] Singal AK, Bataller R, Ahn J, et al. ACG clinical guideline: alcoholic
liver disease. Am J Gastroenterol. 2018;113:175–94.
4. Conclusions [5] Tripodi A, Mannucci PM. The coagulopathy of chronic liver disease. N
Engl J Med. 2011;365:147–56.
The symmetrical or multifocal crescent-shaped ICH in the
[6] Pizzi MA, Alejos DA, Siegel JL, et al. Cerebral venous thrombosis asso-
aforementioned images raises serious doubts about the diag- ciated with intracranial hemorrhage and timing of anticoagulation
nosis of CVT, and it is highly recommended to perform MRV after hemicraniectomy. J Stroke Cerebrovasc Dis. 2016;25:2312–6.
examination to confirm the diagnosis earlier and improve the [7] Coutinho JM, van den Berg R, Zuurbier SM, et al. Small juxtacortical hem-
prognosis. Prompt anticoagulation is crucial and advantageous orrhages in cerebral venous thrombosis. Ann Neurol. 2014;75:908–16.
for patients. [8] Schaller B, Graf R. Cerebral venous infarction: the pathophysiological
concept. Cerebrovasc Dis. 2004;18:179–88.
[9] Afifi K, Bellanger G, Buyck PJ, et al. Features of intracranial hemor-
Author contributions rhage in cerebral venous thrombosis. J Neurol. 2020;267:3292–8.
[10] Fuster D, Samet JH. Alcohol use in patients with chronic liver disease.
Conceptualization: Lingjia Xu. N Engl J Med. 2018;379:1251–61.
Data curation: Lingjia Xu. [11] Saleh T, Matta F, Alali F, et al. Venous thromboembolism with chronic
Investigation: Lingjia Xu, Guoping Fu. liver disease. Am J Med. 2011;124:64–8.
Supervision: Guoping Fu. [12] Ceni E, Mello T, Galli A. Pathogenesis of alcoholic liver disease: role of
Validation: Guoping Fu. oxidative metabolism. World J Gastroenterol. 2014;20:17756–72.
Writing – original draft: Lingjia Xu. [13] Yerke J, Bauer SR, Bass S, et al. Effectiveness of venous thromboembolism
prophylaxis in patients with liver disease. World J Hepatol. 2019;11:379–90.
Writing – review & editing: Lingjia Xu, Guoping Fu.
[14] Schlechter M, Lucey K, Peng TJ, et al. Cashew nut sign: a concave
parenchymal hemorrhage caused by cerebral venous thrombosis.
Stroke. 2023;54:e38–9.
References [15] Schaller B. Physiology of cerebral venous blood flow: from experimen-
[1] Silvis SM, de Sousa DA, Ferro JM, et al. Cerebral venous thrombosis. tal data in animals to normal function in humans. Brain Res Brain Res
Nat Rev Neurol. 2017;13:555–65. Rev. 2004;46:243–60.

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