Chronic subdural hematoma associated with type II and type III Galassi arachnoid cysts: illustrative cases

Bac Thanh Nguyen Department of Neurosurgery, Vietnam Military Medical University, Military Hospital 103, Hanoi, Vietnam; and

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Van Dinh Tran Department of Neurosurgery, Vietnam Military Medical University, Military Hospital 103, Hanoi, Vietnam; and

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Jehan Bista The University of Queensland-Ochsner Clinical School, Jefferson, Louisiana

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Trung Van Trinh Department of Neurosurgery, Vietnam Military Medical University, Military Hospital 103, Hanoi, Vietnam; and

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BACKGROUND

Arachnoid cysts (ACs) are congenital abnormalities that can be located anywhere within the subarachnoid space along the cerebrospinal axis, although they are most often found on the left side in the temporal fossa and sylvian fissure. ACs comprise approximately 1% of all intracranial space-occupying lesions and are considered potential risk factors for subdural hematoma (SDH) in individuals of all age groups who have experienced traumatic brain injury. Although it is uncommon for an intracystic hemorrhage of an AC to occur without evidence of head trauma, it may be more common among children and young adults. Here, the authors present three cases of spontaneous AC intracystic hemorrhage with chronic SDH. Additionally, they provide a thorough review of the existing literature.

OBSERVATIONS

All three patients with AC were adolescent males. In all cases, AC was identified using the Galassi classification (type II or III) and associated with spontaneous intracystic hemorrhage and chronic SDH as seen on imaging.

LESSONS

Spontaneous intracystic hemorrhage is a rare complication and occurs most commonly on the left side. Surgery is the definitive treatment, requiring either craniotomy or burr hole for hematoma evacuation and microsurgical fenestration to drain the cyst into the subarachnoid cisterns.

ABBREVIATIONS

AC = arachnoid cyst; CSF = cerebrospinal fluid; CT = computed tomography; GCS = Glasgow Coma Scale; ICP = intracranial pressure; LOC = loss of consciousness; MRI = magnetic resonance imaging; SDH = subdural hematoma; TBI = traumatic brain injury

BACKGROUND

Arachnoid cysts (ACs) are congenital abnormalities that can be located anywhere within the subarachnoid space along the cerebrospinal axis, although they are most often found on the left side in the temporal fossa and sylvian fissure. ACs comprise approximately 1% of all intracranial space-occupying lesions and are considered potential risk factors for subdural hematoma (SDH) in individuals of all age groups who have experienced traumatic brain injury. Although it is uncommon for an intracystic hemorrhage of an AC to occur without evidence of head trauma, it may be more common among children and young adults. Here, the authors present three cases of spontaneous AC intracystic hemorrhage with chronic SDH. Additionally, they provide a thorough review of the existing literature.

OBSERVATIONS

All three patients with AC were adolescent males. In all cases, AC was identified using the Galassi classification (type II or III) and associated with spontaneous intracystic hemorrhage and chronic SDH as seen on imaging.

LESSONS

Spontaneous intracystic hemorrhage is a rare complication and occurs most commonly on the left side. Surgery is the definitive treatment, requiring either craniotomy or burr hole for hematoma evacuation and microsurgical fenestration to drain the cyst into the subarachnoid cisterns.

ABBREVIATIONS

AC = arachnoid cyst; CSF = cerebrospinal fluid; CT = computed tomography; GCS = Glasgow Coma Scale; ICP = intracranial pressure; LOC = loss of consciousness; MRI = magnetic resonance imaging; SDH = subdural hematoma; TBI = traumatic brain injury

Arachnoid cysts (ACs) are classified as congenital accumulations of cerebrospinal fluid (CSF) that are situated within the arachnoid membrane. These cysts are most often found in the middle cranial fossa. ACs tend to prefer the left side and often do not produce any symptoms but can become symptomatic if they grow sizably or if there is bleeding within the cyst or subdural space.1 Although ACs are typically discovered incidentally, they can sometimes present with clinical signs and symptoms such as macrocephaly in the neonatal period, headaches, hydrocephalus, and epileptic seizures.2

The diagnosis of asymptomatic ACs has increased in recent years due to the extensive utilization of computed tomography (CT) and magnetic resonance imaging (MRI) techniques.3 Head trauma is one of the most significant risk factors for the development of intracystic hemorrhage in ACs and subsequent subdural hematoma (SDH). However, fewer than 182 cases of spontaneous intracystic hemorrhage have been reported in the literature.3,4 In this report, we present illustrative cases of three young patients who experienced spontaneous AC intracystic hemorrhage followed by chronic SDH. Additionally, we provide a thorough review of the existing literature.

Illustrative Cases

Case 1

An 18-year-old male experienced a mild traumatic brain injury (TBI) after a head collision in a traffic accident. During the acute trauma phase, there were no indications of loss of consciousness (LOC) or other severe symptoms. However, 3 months after the incident the patient experienced a worsening headache that became continuous, unresponsive to analgesic treatment, and associated with vomiting. Neurological examination revealed a Glasgow Coma Scale (GCS) score of 15, with no observed changes in pupillary responses, language, or motor function, or signs of epilepsy. After a head CT, the patient’s lesion was identified as a left temporal Galassi type III arachnoid cyst with corresponding chronic SDH (Fig. 1). The patient was urgently admitted to the hospital for microsurgical intervention. The corrective procedure involved a left pterional craniotomy, drainage of the hematoma, and microsurgical arachnoid cyst fenestration. This surgical approach aimed to establish communication between the cyst and interpeduncular cistern. The patient’s headache improved during the immediate postoperative period. He was discharged after 2 weeks with no residual deficits.

FIG. 1
FIG. 1

Left: Preoperative axial head CT with contrast showing a left temporal Galassi type III AC and chronic left SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and SDH.

Case 2

A 14-year-old male presented with worsening headache that started 2 days prior to admission without associated trauma. He then came to the hospital for further evaluation. On examination his GCS score was 15 with no pupillary, language, or motor changes. Other than headache, he was asymptomatic, with no signs of epilepsy. An initial CT head identified a left temporal Galassi type II AC with corresponding chronic SDH (Fig. 2). The patient underwent a left pterional craniotomy, hematoma drainage, and microsurgical AC fenestration. The patient’s headache improved in the immediate postoperative period, and he was discharged after 2 weeks with good outcomes.

FIG. 2
FIG. 2

Left: Preoperative axial head CT with contrast showing a left temporal Galassi type II AC with chronic left SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and SDH.

Case 3

A 21-year-old male experienced a mild TBI after a head collision in a traffic accident. During the acute trauma phase, there were no indications of LOC or other severe symptoms. However, 2 months after the incident the patient’s headache intensified and became continuous, unresponsive to analgesic treatment, and accompanied by vomiting. Neurological examination revealed a GCS score of 15, with no pupillary, motor, language, or epileptic signs. An initial CT head identified the lesion as a left temporal Galassi type II AC with associated chronic SDH (Fig. 3). The patient was placed under local anesthesia, and a burr hole was created for hematoma evacuation. Upon performing the procedure, the brain promptly re-expanded. The AC was not excised during surgery, and a subdural drain was not required for additional drainage. The patient was discharged after 10 days with good postoperative outcomes.

FIG. 3
FIG. 3

Left: Preoperative axial head CT with contrast showing a left temporal Galassi type II AC w/associated left chronic SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and chronic SDH.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

All participants included in our study were adolescent males. Previous studies have reported the prevalence of AC in the adult population to fall between 0.23% and 2.43%. In the pediatric population (those 18 years or younger), the estimated prevalence is 2.6%. Notably, both adult and pediatric populations exhibit a higher prevalence of AC among male patients.5

SDHs are common, with an annual incidence of 13 to 14 cases per 100,000 person-years, and many cases remain undiagnosed. Most SDHs are associated with head trauma, wherein deceleration forces cause the rupture of bridging veins within the subdural space. Spontaneous SDHs are less frequent, accounting for 2% to 5% of atraumatic cases. The etiology of these hematomas, excluding those associated with arteriovenous malformations or fistulae, is not fully understood. However, they have been observed in patients with tendency to bleed due to hematological disorders, malignancies, anticoagulation therapy, hypertension from preeclampsia, infections, and hypervitaminosis. Rupture of cortical arteries at their adhesion sites with dura mater has also been reported. Spontaneous SDHs may occur during Valsalva maneuvers (e.g., coughing, straining, or weight training), or in association with intracranial hypotension caused by CSF leakage, excessive shunt drainage, exercise, or dehydration.6

In adults, ACs are frequently encountered as an incidental finding on imaging. Although ACs may present with symptoms during childhood, they often remain asymptomatic until adulthood. According to one study, the prevalence of ACs was found to be 1.4% in patients who underwent MRI. Only a small percentage (5.3%) of the cysts were symptomatic.7 Despite being considered benign, ACs can give rise to complications such as SDH, hygroma, and intracystic hemorrhage. These complications can occur spontaneously or due to minor head trauma. The bleeding is believed to be caused by the absence of structural support in the veins surrounding the ACs. This hypothesis is supported by the vascular nature of the cystic membrane as well as the presence of bridging veins traversing the cyst. Notably, the reduced compliance of the cyst compared to normal brain tissue may contribute to bridging vein rupture, particularly during instances of increased intracranial pressure (ICP).2 Gosalakkal8 identified that ACs within the middle cranial fossa were present in 2.43% of patients diagnosed with chronic SDHs or hygromas. Patients with chronic SDH had five times the prevalence of ACs when compared with controls.8

In our study, all three ACs bled with associated chronic SDH. All cases were associated with mild head trauma and large cyst size according to the Galassi classification (types II and III). These cases demonstrate the influence of mild TBI as a risk factor for subsequent AC rupture. A case-control study revealed that recent trauma within the past 30 days might elevate the risk of AC rupture by as much as 26.5-fold. Additionally, cyst size greater than 50 mm was identified as another risk factor for rupture.9

Clinical signs or symptoms often seen in the setting of ACs include epilepsy, attention-deficit/hyperactivity disorder, speech or developmental delay, signs of obstructive hydrocephalus, aphasia, increased ICP, headaches, and vomiting.5,8 In our study, none of the patients experienced significant symptoms other than headache or vomiting prior to hemorrhage.

The most effective treatment approach for chronic SDH associated with AC remains a topic of debate. Treatment for ACs is based on their location and presence of symptoms. Symptomatic cysts may require surgical intervention, but most neurosurgeons do not recommend treating asymptomatic cysts. ACs that are linked to subdural or epidural hematomas may also resolve on their own. In cases in which conservative measures are ineffective, neurosurgical intervention can be considered for AC management. Surgical options include shunting procedures, craniotomy with cyst fenestration, use of burr holes, and endoscope-assisted cyst fenestration into the CSF spaces.2,5,9

Burr hole and craniotomy are the primary surgical techniques employed for draining chronic SDH.

In certain cases, surgeons may opt not to treat ACs when performing burr hole procedures. Others may choose to address the cyst through various methods including partial cyst removal or microsurgical cyst fenestration toward the interpeduncular or optic chiasmatic cisterns.5,9 Wu et al.4 conducted a comprehensive review focusing on AC-associated chronic SDH in both adult and pediatric patients (n = 182). Their study revealed recurrence rates of 8.2% with burr hole procedures and 1.5% with craniotomy.4

Some authors have proposed the use of burr holes without any manipulation of the AC as the initial preferred approach for symptomatic patients and for selected instances of recurrence.4 Other authors have suggested that the optimal treatment modality for patients with AC with associated hemorrhage involves hematoma evacuation and radical marsupialization.10 After cyst rupture and hemorrhage, endoscopic surgery presents technical challenges due to limited visibility and orientation within the cyst and surrounding membranes. These factors hinder the safe execution of fenestration procedures.9

In the first two cases, we assumed that the cyst wall had been opened because the contents within the AC and chronic SDH were of similar density. In both cases a frontotemporal craniotomy was performed, and the membranes of the hematoma and AC were completely removed to achieve complete evacuation. In the third case, a burr hole was made to evacuate the chronic SDH because the boundary between the cyst wall and hematoma was visible.

Lessons

AC with spontaneous intracystic hemorrhage and corresponding chronic SDH is a relatively rare condition. Surgery is considered the most effective treatment approach. In addition, evacuating chronic SDHs, microsurgical fenestration and membranectomy are recommended to prevent recurrence.

Author Contributions

Conception and design: Nguyen, Trinh. Acquisition of data: Nguyen, Trinh. Analysis and interpretation of data: Nguyen, Trinh. Drafting the article: all authors. Critically revising the article: Bista, Nguyen, Trinh. Reviewed submitted version of manuscript: Bista, Nguyen, Trinh. Approved the final version of the manuscript on behalf of all authors: Bista. Statistical analysis: Trinh. Administrative/technical/material support: Tran, Trinh. Study supervision: Nguyen.

References

  • 1

    Al-Holou WN, Yew AY, Boomsaad ZE, Garton HJL, Muraszko KM, Maher CO. Prevalence and natural history of arachnoid cysts in children. J Neurosurg Pediatr. 2010;5(6):578585.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    Bilginer B, Önal MB, Oğuz KK, Akalan N. Arachnoid cyst associated with subdural hematoma: report of three cases and review of the literature. Childs Nerv Syst. 2009;25(1):119124.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Aydogmus E, Hicdonmez T. Spontaneous intracystic haemorrhage of an arachnoid cyst associated with a subacute subdural hematoma: a case report and literature review. Turk Neurosurg. 2019;29(6):940944.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Wu X, Li G, Zhao J, Zhu X, Zhang Y, Hou K. Arachnoid cyst-associated chronic subdural hematoma: report of 14 cases and a systematic literature review. World Neurosurg. 2018;109:e118e130.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Gregori F, Colistra D, Mancarella C, Chiarella V, Marotta N, Domenicucci M. Arachnoid cyst in young soccer players complicated by chronic subdural hematoma: personal experience and review of the literature. Acta Neurol Belg. 2020;120(2):235246.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Hall A, White MAJ, Myles L. Spontaneous subdural haemorrhage from an arachnoid cyst: a case report and literature review. Br J Neurosurg. 2017;31(5):607610.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Offiah C, St Clair Forbes W, Thorne J. Non-haemorrhagic subdural collection complicating rupture of a middle cranial fossa arachnoid cyst. Br J Radiol. 2006;79(937):7982.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Gosalakkal JA. Intracranial arachnoid cysts in children: a review of pathogenesis, clinical features, and management. Pediatr Neurol. 2002;26(2):9398.

  • 9

    Furtado LMF, Costa Val Filho JA, Ferreira RI, Mariano IGGF. Intracranial arachnoid cyst rupture after mild TBI in children: have we underestimated this risk? BMJ Case Rep. 2019;12(4):e228790.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Chandra VV, Prasad BC, Subramanium CS, Kumar R. Spontaneous intracystic hemorrhage complicating an intracranial arachnoid cyst. J Neurosci Rural Pract. 2015;6(4):629630.

    • PubMed
    • Search Google Scholar
    • Export Citation
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  • Expand
  • FIG. 1

    Left: Preoperative axial head CT with contrast showing a left temporal Galassi type III AC and chronic left SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and SDH.

  • FIG. 2

    Left: Preoperative axial head CT with contrast showing a left temporal Galassi type II AC with chronic left SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and SDH.

  • FIG. 3

    Left: Preoperative axial head CT with contrast showing a left temporal Galassi type II AC w/associated left chronic SDH. Right: Postoperative axial head CT without contrast demonstrating total resection of the AC and chronic SDH.

  • 1

    Al-Holou WN, Yew AY, Boomsaad ZE, Garton HJL, Muraszko KM, Maher CO. Prevalence and natural history of arachnoid cysts in children. J Neurosurg Pediatr. 2010;5(6):578585.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2

    Bilginer B, Önal MB, Oğuz KK, Akalan N. Arachnoid cyst associated with subdural hematoma: report of three cases and review of the literature. Childs Nerv Syst. 2009;25(1):119124.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Aydogmus E, Hicdonmez T. Spontaneous intracystic haemorrhage of an arachnoid cyst associated with a subacute subdural hematoma: a case report and literature review. Turk Neurosurg. 2019;29(6):940944.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Wu X, Li G, Zhao J, Zhu X, Zhang Y, Hou K. Arachnoid cyst-associated chronic subdural hematoma: report of 14 cases and a systematic literature review. World Neurosurg. 2018;109:e118e130.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Gregori F, Colistra D, Mancarella C, Chiarella V, Marotta N, Domenicucci M. Arachnoid cyst in young soccer players complicated by chronic subdural hematoma: personal experience and review of the literature. Acta Neurol Belg. 2020;120(2):235246.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Hall A, White MAJ, Myles L. Spontaneous subdural haemorrhage from an arachnoid cyst: a case report and literature review. Br J Neurosurg. 2017;31(5):607610.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Offiah C, St Clair Forbes W, Thorne J. Non-haemorrhagic subdural collection complicating rupture of a middle cranial fossa arachnoid cyst. Br J Radiol. 2006;79(937):7982.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Gosalakkal JA. Intracranial arachnoid cysts in children: a review of pathogenesis, clinical features, and management. Pediatr Neurol. 2002;26(2):9398.

  • 9

    Furtado LMF, Costa Val Filho JA, Ferreira RI, Mariano IGGF. Intracranial arachnoid cyst rupture after mild TBI in children: have we underestimated this risk? BMJ Case Rep. 2019;12(4):e228790.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Chandra VV, Prasad BC, Subramanium CS, Kumar R. Spontaneous intracystic hemorrhage complicating an intracranial arachnoid cyst. J Neurosci Rural Pract. 2015;6(4):629630.

    • PubMed
    • Search Google Scholar
    • Export Citation

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