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. 2020 Sep 1;12(9):e10183. doi: 10.7759/cureus.10183

Hemorrhagic Vestibular Schwannoma: Case Report and Literature Review of Incidence and Risk Factors

Taha Shahbazi 1, Mohammadmahdi Sabahi 2, Mahdi Arjipour 3,, Badih Adada 4, Hamid Borghei-Razavi 5
Editors: Alexander Muacevic, John R Adler
PMCID: PMC7529481  PMID: 33029463

Abstract

Hemorrhagic vestibular schwannoma (HVS) consisting of acute intratumoral and subarachnoid hemorrhage presents with acute nausea, vomiting, facial numbness, headache, loss of consciousness, and significant functional impairment of the facial and vestibulocochlear nerves. The current case is of a 31-year-old man who was presented with acute left lateral suboccipital headache, vomiting, ataxia, and loss of consciousness. Brain CT revealed a large iso-intense lesion with internal hematoma at the left cerebellopontine angle in association with internal acoustic canal dilation. In addition, MRI confirmed a 32 x 25 x 26 mm vestibular schwannoma (VS) with 20 x 15 x 5 mm intratumoral hematoma. The patient had undergone left lateral suboccipital craniotomy and microscopic tumor resection. Pathological findings revealed that his lesions were VS. The average incidence of HVS is around 2.15 cases per year worldwide. Therefore, HVS incidence in proportion to VS is very low and consequently rare.

Keywords: intratumoral hemorrhage, subarachnoid hemorrhage, vestibular schwannoma, acoustic neuroma, hemorrhage

Introduction

Vestibular schwannoma (VS), also called acoustic neuroma, is a benign tumor of the vestibulocochlear nerve that has an incidence of 11-13 cases per million every year. It arises from Schwann cells at the Obersteiner-Redlich zone [1-3].

Hemorrhagic VS (HVS) is a rare phenomenon. Only limited cases are reported and there has been no definite estimation about HVS incidence until now. When significant HVS occurs, patients may experience acute neurological defects [4]. Considered risk factors include huge tumor size, cystic development, hypointense parts in T2-weighted magnetic resonance (MR) signal, hemosiderin deposition due to intratumoral hemorrhage (ITH), and a history of anticoagulant therapy [5,6].

The treatment is based on surgery, and radiation therapy (RT) and observation are other choices of treatment. There are three standard surgical approaches: retromastoid suboccipital (retrosigmoid), translabyrinthine, and middle fossa approach (MFA). Although surgery is the treatment of choice for VS, there are many different RT techniques including single-session stereotactic radiosurgery (SRS), fractionated conventional RT, fractionated stereotactic RT (FSRT), and proton therapy [7-10].

Herein, we report a case of a patient who was admitted with HVS. Some similar cases have also been reviewed as literature review.

Case presentation

All procedures performed in studies involving human participants were in accordance with the ethical standards (code: IR.UMSHA.REC.1399.388) and the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. Of note, informed consent was obtained from the patient in order to use his information for the case presentation. A 31-year-old shepherd man was referred to our center due to loss of consciousness. He was able to communicate and explained his experiences of a sudden severe headache in the left lateral suboccipital (retroauricular) region, as well as left hemi-facial numbness, nausea, and vomiting. After a while, he developed disequilibrium and loss of consciousness instantly after walking a few steps. Neurological examination showed he had facial paresis (House-Brackmann grade II), vertigo, and ataxia, whereas other examinations were normal.

His past medical history revealed he had a history of appendectomy 30 months before current admission. He had also developed deep vein thrombosis and pulmonary emboli 18 months ago and had used aspirin after thrombolytic therapy. Moreover, he had a history of surgical removal of lung hydatid cyst 12 months ago as well as a history of headache for three years, which was intractable to any treatment. His prothrombin time (PT), partial thromboplastin time (PTT), and international normalized ratio (INR) were normal, and no abnormal findings were reported in lab results.

Brain CT revealed extra-axial lesion at the left cerebellopontine angle (CPA) region with an intralesion hematoma that expanded tumor volume (Figure 1A). The left internal acoustic canal (IAC) was dilated in comparison with the right side. The brain stem and fourth ventricle were compressed with the mass effect of hemorrhagic tumor. There was some ventriculomegaly but no periventricular edema and hydrocephalus.

Figure 1. Patient's Preoperative Imaging.

Figure 1

(A) Brain CT shows an extra-axial lesion at the left CPA region with intralesion hematoma. (B and C) Axial T1- and T2 -weighted images, respectively, show extra-axial lesion, which was hypointense on T1-weighted images and hyperintense on T2-weighted images. Intralesional hematoma is iso- and hypointense on T1- and T2-weighted images, respectively. (D) Sagittal T2-weighted image. (E) Axial MRI source image. (F and G) Contrast T1-weighted axial and sagittal images show homogenous avid tumor enhancement. (H) Contrast CT show postoperation changes and tumor resection with retrosigmoid approach.

In the MRI, 32 x 25 x 26 mm VS at the left CPA with extension to IAC was seen. The lesion was hypo- and hyperintense on T1-weighted image (T1WI) and T2-weighted image (T2WI), respectively (Figures 1B-1D) and enhanced dens homogenous on contrast T1WI (Figures 1F, 1G). Intralesion hematoma measuring 20 x 15 x 5 mm was iso- and hypointense on T1WI and T2WI, respectively, without T1WI enhancement contrast (Figure 1).

The patient was operated at semi-lateral supine position with left lateral suboccipital craniotomy and microscopic retrosigmoid approach. Intratumoral hematoma was seen during tumor resection. The tumor was totally resected, and the facial nerve was preserved. The patient’s postoperation course was uneventful and without any new neurological deficit (Figures 2A, 2B).

Figure 2. Lesion Microscopic View.

Figure 2

(A) Low power field. (B) High power field microscopic view shows vestibular schwannoma. Antoni A and B patterns are seen in the collagenous background. In the hypercellular Antoni A areas, intersecting fascicles consisting of spindle cells with buckled nuclei are seen. These cells form Verocay bodies with nuclear palisading, making alternating bands of nuclear and anuclear areas. In the hypocellular Antoni B areas, the prominent myxoid extracellular matrix takes the spindle cells apart.

In the hematoxylin and eosin (H & E) staining, hypercellular (Antoni A) and hypocellular (Antoni B) areas are seen. Dense Antoni A areas consisting of interlacing bundles of spindle cells with oval nuclei, eosinophilic cytoplasm, and indistinct cytoplasmic borders are also seen. Hypocellular dense Antoni B areas are composed of haphazardly arranged spindle cells in loose myxoid collagen fibers. Blood vessels with hyalinized walls were visible.

Discussion

The VS is an uncommon intracranial tumor and frequently presents with chronic hearing loss, headache, tinnitus, disequilibrium, and facial numbness, whereas acute overt hemorrhage including subarachnoid hemorrhage (SAH) and ITH as its first presentation is quite rare [11].

Of all intracranial hemorrhages, 1-11% belong to hemorrhagic brain tumors. SAH that arises from brain tumors accounts for 0.4% of all cases of SAH. Furthermore, 1.7- 10% of brain tumors cause intracerebral hemorrhage [12]. ITH generally occurs in 11% of all cranial tumors, and its occurrence in glioblastoma multiforme, pituitary adenomas, choriocarcinomas, oligodendrogliomas, choroid plexus papillomas, and meningiomas is also prevalent.

According to Mathkour et al.’s literature review, there were only 48 cases of VS secondary to ITH, whereas in accordance with our review of the literature (Table 1), there were 97 HVS cases since 1974 including our case [13].

Table 1. Reported Hemorrhagic Vestibular Schwannoma.

Abbreviations: AF, arterial fibrillation; CABG, coronary artery bypass grafting; CAD, coronary artery disease; CN, cranial nerve; CSF, cerebrospinal fluid; EVD, external ventricular drain; F, female; GFAP, glial fibrillary acidic protein; HB, House-Brackmann grade; HD, hearing disturbance; HTN, hypertension; ITH, intratumoral hemorrhage; IVH, intraventricular hemorrhage; LAFT, lack of access to full text; M, male; MI, myocardial infarction; NR, not reported; NS, not otherwise specified; N/V, nausea/vomiting; PMH, past medical history; POC, postoperative conditions; RF, risk factor; RM, retromastoid; RS, retrosigmoid; SAH, subarachnoid hemorrhage; SO, suboccipital craniotomy; SRS, stereotactic radiosurgery; TBI, traumatic brain injury; TL, translabyrinthine; VPS, ventriculoperitoneal shunt; VS, vestibular schwannoma

Remarks Outcome, recurrence Tumor characteristics Treatment Pre/postsurgery HB grade SAH Size (cm) Clinical presentation Age(years)/Sex Case number
History of HTN and pregnancy Death ITH/macroscopic characteristics: gray, firm, and not attached to the tentorium Conservative NR/NR Yes 2 x 3 x 3.5 N/V, facial hemiparesis, progressive slowed mental reactions 64/F 1
Bloody CSF Good, NR ITH Elective Surgery NR/NR Yes Large Headache, N/V, vertigo 21/F 2
POC: persistent ataxia, facial paresis, and HD Good, NR No ITH SO NR/NR Yes 4 Headache, HD, facial numbness and paresthesia, nystagmus, decrease corneal reflex, hypoesthesia and hypoalgesia of left trigeminal nerve 54/F 3
NR Death No ITH Conservative NR/NR Yes NR NR 64/M 4
LAFT Good, NR No ITH LAFT NR/NR Yes NR Headache, ataxia 54/M 5
Hydrocephalus and VPS Death, ITH Surgery NS NR/NR No NR Ataxia, facial hemiparesis, headache, hydrocephalus, altered mental status 71/F 6
PMH: deafness, ataxia and sensory change over the left trigeminal territory; job: farmer Good, NR No ITH Surgery NS NR/NR Yes 3 Ataxia, HD, facial hypoesthesia 66/M 7
Xanthochromic CSF; PMH: tinnitus for 2 years, unsteadiness of balance with vertigo/no facial palsy Good, NR ITH SO NR/NR canal. Yes 4 Headache, nystagmus, dizziness, otalgia, N/V, HD, absent ice-water caloric response, decreased corneal reflex, decreased right external auditory sensation 61/M 8
Subdural hemorrhage Good, NR ITH SO NR/NR Yes 5 Ataxia, facial hypoesthesia, facial hemiparesis, headache, N/V, papilledema 49/M 9
RF: pregnancy Good, NR ITH LAFT NR/NR No NR Headache, dizziness, swallowing difficulty 33/F 10
LAFT Good, NR ITH LAFT NR/NR Yes NR Tinnitus, HD 54/M 11
NR Good, NR ITH Surgery NS NR/NR NR 4 Headache, HD, tinnitus 33/F 12
LAFT Good, NR No ITH LAFT NR/NR Yes NR Headache, N/V 32/M 13
LAFT Good, NR ITH LAFT NR/NR Yes 3 Headache 61/F 14
Anticoagulation valve replacement Death ITH LAFT LAFT LAFT Large Headache, diplopia, facial hypoesthesia, unsteadiness 58/M 15
RF: heavy weight lifting, head injury; POC: facial nerve was preserved. Good, NR ITH/no cystic tumor SO, TL NR/NR Yes 3.2 Headache, HD, facial hemiparesis, otalgia, N/V, vertigo, sweating, nystagmus, taste perception was absent on the left side 19/M 16
Physical exercise/PMH: Grave's disease, HD, and tinnitus Good, NR ITH Surgery NR/NR Yes NR Headache, N/V, facial sensory change, loss of consciousness, stiff neck, Brun's nystagmus, absence of right corneal reflex 47/F 17
NR Good, NR ITH RS NR/NR No NR Facial sensory change, HD 37/F 18
RF: HTN Good, NR ITH SO NR/NR No NR Headache, N/V, diplopia 60/M 19
NR Good, NR ITH NR NR/NR No 5 Headache, HD, vomiting, diplopia, facial paresthesia, nystagmus 42/M 20
Headache, cysts filled with dark red or xanthochromic fluid Good, NR ITH, cystic tumor Surgery NS NR/II No 5 Headache, HD, ataxia, trigeminal and facial nerve palsies 56/M 21
NR Good, NR ITH Surgery NR/NR No 3 Headache, tinnitus, HD 39/M 22
Postoperation facial weakness and HD Good, NR ITH SO NR/NR No 4 Ataxia, dizziness, headache, N/V, vertigo, nystagmus, decreased corneal reflex, stiff neck 65/F 23
Minor head injury; after surgery, slight left facial weakness and hearing loss persisted Good, NR ITH/tumor had multiple cysts which consisted of xanthochromic and dark brown elements SO NR/NR No 3 Ataxia, facial hemiparesis, HD, Brun’s nystagmus 62/F 24
NR Good, NR ITH NR NR/NR No 5.4 Ataxia, HA, HD 41/M 25
NR NR, NR NR SO NR/NR Yes NR Gait disturbance, Headache HD, N/V, facial numbness 70/M 26
NR NR, NR ITH SO NR/NR Yes 3 Progressively worsening left suboccipital neck pain, ataxia, facial paresis 42/F 27
Gag reflexes were depressed, no facial weakness or sensory loss was present NR, NR ITH Surgery NR/NR No 2,3 HD, increasing gait Instability, dizziness 66/F 28
NR NR, NR NR Surgery NR/NR Yes 4 Severe neurological impairment due to cerebral compression, persistent diplopia, headache, HD 23/M 29
PMH: persistent left tinnitus and left hearing loss, feeling of tingling appeared around the left tongue and left lip, vertigo Good, NR ITH Posterior subcraniotomy NR/NR No 4 x 3 x 3 HD, ataxia, unilateral headache and trigeminal symptom, facial paresthesia, decreased perception of the third branch (only around the lips), decreased taste sensation 46/F 30
NR Good, NR ITH RS NR/NR No 3 Ataxia, facial hemiparesis, HD, N/V 63/F 31
NR Good, NR ITH RS NR/NR No 3.8 Ataxia, headache, HD, N/V 45/F 32
NR Good, NR ITH RS NR/NR No 2.8 Facial hypoesthesia, headache, HD 31/M 33
NR Good, NR ITH RS NR/NR No 3 Ataxia, headache, HD 54/M 34
NR Good, NR NR RS NR/NR   3 Headache, nystagmus 28/M 35
CN VII to XII paralysis Good, NR ITH LAFT LAFT LAFT 2.6 Tinnitus, ataxia, facial paralysis, hoarseness 65/F 36
NR NR, NR ITH NR NR/NR No NR HD, dizziness, absent gag and left corneal reflexes NR 37
NR Good, NR NR Surgery NR/NR No Large tumor (>2 cm) HD, facial hemiparesis, cerebellar symptomatology, N/V  70/M 38
None of the other five patients (41-45) had an acute onset; none of the patients had systemic HTN Good, NR ITH/cystic tumor RM NR/NR No 3 Tinnitus, HD, headache, facial paresis, ataxia 65/F 39
Death ITH/cystic tumor RM NR/NR Yes 4.2 NR 35/F 40
Good, NR ITH/cystic tumor RM NR/NR No 4.0 NR 30/F 41
Good, NR ITH/cystic tumor RM NR/NR No 6.0 NR 32/M 42
Good, NR ITH/cystic tumor RM NR/NR No 5.0 NR 43/F 43
Good, NR ITH/cystic tumor RM NR/NR No 3.8 NR 72/M 44
Good, NR ITH/cystic tumor RM NR/NR No 5.2 NR 44/F 45
PMH: HD on the left and tinnitus for 20 years and headache from 1 week prior to admission Good, NR ITH SO NR/NR No 3.5 Left HD, gaze nystagmus toward the right 66/F 46
Aspirin for his pain Good, NR ITH SO NR/NR   2.5 HD, intermittent tinnitus, mild intermittent left-sided headaches 36/M 47
No tumor recurrence after 6 years Good, No ITH SO NR/II No 4 Ataxia, facial hypoesthesia, Headache, HD, N/V, tinnitus, hydrocephalus 35/F 48
NR Good, No ITH RS NR/NR   NR HD, headache, tinnitus, no caloric response 25/F 49
PMH: HTN, hypercholesterolemia Good, No ITH TL NR/NR No 1.9 Dizziness, HD, N/V, tinnitus 55/M 50
NR Good, No ITH/cystic tumor RS VI/VI No 2 Facial hemiparesis, HD 73/F 51
NR Good, No ITH TL I/I No 2.6 Diplopia, facial hypoesthesia, Headache, HD, N/V, otalgia 52/F 52
NR Good, No ITH/cystic tumor TL/RS NR/NR No 3.6 Ataxia, facial hypoesthesia, Headache, HD, otalgia 41/F 53
Recurrent IVH and SAH Good, No NR NR NR/NR Yes 1 Headache facial hemiparesis, N/V, neck pain 15/F 54
LAFT Good, NR ITH/hypervascularized tumor LAFT LAFT LAFT LAFT Several CN palsies, Decreased level of consciousness 38/M 55
NR NR, NR ITH/cystic tumor Surgery NS NR/NR NR 4.4 HD, gait disturbance 19/M 56
NR NR, NR ITH/cystic tumor Surgery NS NR/NR NR 4.5 HD, headache, gait disturbance 64/F 57
Oral anticoagulation treatment Good, NR ITH/positive S-100 protein Surgery NS NR/NR Yes 1.5 Dizziness, facial hemiparesis, headache, HD, vertigo, N/V 49/M 58
PMH: dizziness, HTN, and AF treated by calcium channel blocker and anti-vitamin K were reported Death ITH None NR/NR NR 3 Diplopia, dysphonia, facial hemiparesis, Headache, HD, CN III, V, VI, VII, IX, X, XI deficit 73/F 59
LAFT Good, NR LAFT LAFT NR/NR No NR Headache, HD, facial hemiparesis 68/M 60
PMH: chronic right-sided deafness, right facial nerve paresis, and mild ataxia treated using low-dose aspirin Good, NR ITH SRS NR/NR No 2 HD, facial palsy 55/M  61
PMH: stereotactic radiation therapy for VS due to her cardiac condition Death ITH/cystic tumor/highly vascularized tumor surgery NR/NR NR NR Hemiparesis, loss of consciousness 72/F 62
NR Good, NR No ITH/no cystic tumor RS NR/NR NR 1.6 HD 41/F 63
NR Good, NR ITH/cystic tumor RS I/NR NR 3 Facial hypoesthesia, headache, N/V 48/F 64
NR Good, NR No cystic tumor TL II/NR NR 3.6 Ataxia, headache 47/F 65
NR Good, NR Cystic tumor RS II/NR NR 4 Dizziness, headache, N/V 26/F 66
NR Good, NR ITH/no cystic tumor RS II/NR NR 1.8 Facial hemiparesis, headache 68/M 67
NR Death No cystic tumor VPS I/NR NR 2.3 Diplopia, headache, neck pain 66/M 68
PMH: rheumatoid arthritis treated with methotrexate (thrombocyte count remained normal) Good, No ITH VPS, RS NR/V NR NR Ataxia, headache, HD, papilledema, hydrocephalus 59/F 69
NR Good, NR No ITH SO NR/NR Yes NR HD 18/F 70
NR Good, NR ITH RS II/V No 2.5 Headache, vertigo, N/V 66/M 71
PMH: HTN, hyperlipidemia, CAD with MI, and paroxysmal AF, CABG mitral valve replacement, Warfarin, mitral valve replacement, and prostatectomy Death ITH/cystic tumor Conservative VI/VI No 3.2 Ataxia, facial hemiparesis, facial hypoesthesia, HD 69/M 72
Hearing did not improve and he developed mild facial paralysis Good, NR ITH RS III/III NR 3.1 Ataxia, sudden onset Headache, HD, nausea, tinnitus 15/M 73
Cerebral angiography was normal Good, NR ITH SO NR/NR NR 3 Facial hemiparesis, headache, HD 65/F 74
POC: improved hearing; no recurrence Good, No ITH Observation I/I NR 0.4 Ataxia, vertigo, tinnitus 83/NR 75
POC: facial nerve sacrifice; no recurrence Good, No ITH Surgery I/VI NR 3.5 HD, headache, ataxia 39/NR 76
POC: improved gait, vertigo; no recurrence Good, No ITH Surgery I/II NR 2.0 HD, headache, ataxia, vertigo, trigeminal weakness 66/NR 77
POC: improved gait, vertigo, diplopia; no recurrence Good, No ITH Surgery VI/II NR 3.5 HD, headache, facial Weakness, vertigo, diplopia 68/NR 78
No recurrence Good, No ITH Surgery I/NR NR 3.1 HD, ataxia, headache, N/V, hydrocephalus 72/NR 79
No recurrence Good, No ITH Surgery VI/NR NR 2.4 HD, headache, facial Weakness, vertigo, tinnitus 61/NR 80
PMH; HTN, hypercholesterolemia, thrombosis of the right carotid artery, acute MI, total knee replacement, thoracic and lumbar fractures, diabetic retinopathy, aortic valve replacement, CABG, and postoperative AF, extensive medications, which included anti‑coagulants Death ITH/positive S-100, and a low mitotic activity (MIB1) in tumor areas, GFAP staining was negative, cystic tumor RS VI/NR No NR Facial hemiparesis, headache, HD 76/F 81
RF: HTN Good, No ITH/no cystic tumor RS NR/II NR 2.4 Facial hemiparesis, dizziness, Headache, N/V 66/M 82
RF: HTN anticoagulant (INR 2.8)/requiring VPS Good, No ITH/no cystic tumor EVD, then TL NR/VI NR 4.2 Ataxia, dizziness, facial hemiparesis, Headache, HD, Hydrocephalus 39/M 83
RF: HTN, anticoagulant (INR 2.3) Good, No ITH/cystic tumor RS IV/I NR 3.1 Dizziness, facial hemiparesis, facial hypoesthesia, headache, HD, hydrocephalus 68/M 84
RF: HTN requiring VPS Good, No ITH/no cystic tumor EVD, then RS NR/V NR 3.5 Ataxia, dizziness, facial hemiparesis, facial hypoesthesia, headache, HD, hydrocephalus 72/F 85
NR Good, No ITH/cystic tumor TL IV/IV NR 2.4 Dizziness, facial hemiparesis, headache, HD 61/M 86
NR Good, Yes ITH/no cystic tumor RS III/II Yes 4.1 Ataxia, diplopia, facial hemiparesis, headache, HD, N/V 62/F 87
NR Good, No ITH/no cystic tumor RS III/II No 5.1 Headache, HD, N/V, mental deterioration, hydrocephalus 58/F 88
NR Good, No ITH/no cystic tumor RS II/II No 3 Ataxia, facial paresis, headache, HD 65/M 89
PMH: HTN Death ITH/no cystic tumor RS III/II Yes 3.2 Facial hemiparesis, facial hypoesthesia, HD, hydrocephalus 48/M 90
NR Good, No ITH/no cystic tumor RS III/II Yes 5.1 Diplopia, facial hemiparesis, headache, HD, N/V, hydrocephalus 56/M 91
NR Good, NR ITH RS II/III No 3.9 HD, facial paralysis, tinnitus 42/F 92
PMH: HTN Good, NR ITH RS II/III No 4.3 HD, dizziness, choke, hydrocephalus 71/F  93
PMH: HTN, dyslipidemia, congestive heart failure and AF, taking warfarin, mild TBI; POC: no confusion, no hydrocephalus Good, No Intralesional hemorrhage VPS IV-VI/II-VI NR 3 Hyperacusis, confusion spatial and temporal disorientation, facial palsy, mild hydrocephalus 74/M 94
Postoperation persistent ataxia; PMH: Hodgkin’s lymphoma and gradual hearing loss Good, NR ITH NS IV/IV NR 3 Facial hemiparesis, facial hypoesthesia, headache, HD, tinnitus, ataxia 30/F 95
Crocodile tears 1 year after surgery PMH: Meniere’s disease Good, No ITH RS II/I No 4.1 Facial hemiparesis, facial hypoesthesia, Headache, HD, N/V, rotational nystagmus, dizziness 40/M 96
PMH: headache for 3 years, appendicitis, deep vein thrombosis, hydatid cyst operation Good, No ITH/no cystic tumor RS II/III No 3.2 Loss of consciousness, headache, facial numbness, N/V, disequilibrium, facial paresis 31/M 97

Based on the reported cases, the average incidence of HVS worldwide is approximately only 2.15 cases per year; therefore, HVS is a quite rare entity among VS tumors.

Some studies have reported hemorrhagic cystic VS (CVS) and therefore it can be considered as a risk factor for HVS (Table 1). Although our case’s tumor was not of cystic type, CVS has been characterized with faster expansion rate than the solid ones, rapid nerve involvement, and development of variable symptoms. CVS is more commonly demonstrated with fluid-fluid levels and hemosiderin deposition on imaging. Moreover, it has been argued that ITH can result in the formation of the cyst [14].

It is assumed that anticoagulant therapy could be a potential risk factor for HVS. In accordance with other reports, our case had a history of anticoagulant (aspirin) therapy [15,16].

Our patient had high physical activities and he was a shepherd. The patients reported by others had a history of pregnancy, farming, heavy weight lifting, strenuous exercise, and hypertension [17-20]. As a result, these items can also be considered as potential risk factors for HVS.

Conclusions

Micro-ITH may happen more commonly in VS, but clinically significant hemorrhage has a very rare occurrence. HVS risk factors consist of huge tumor size, cystic development, history of anticoagulant therapy, and strenuous activities.

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The authors have declared that no competing interests exist.

Human Ethics

Consent was obtained by all participants in this study. Hamadan University of Medical Science issued approval IR.UMSHA.REC.1399.388. All procedures performed in studies involving human participants were in accordance with the ethical standards (code: IR.UMSHA.REC.1398.982) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards

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