Remote Cerebellar Hemorrhage due to Cerebrospinal Fluid Leakage or Meningitis after Spinal Surgery: Case Report

A 67 year old man presented with low back pain and gradually aggravated paresthesia of left leg caused by spinal stenosis (the fourth and fifth lumbar, the fifth lumbar and the first sacrum). He reported that the symptoms started 1 year ago and he had underwent discectomy (the fifth lumbar and the first sacrum) 10 year ago and vertebroplasty (the second lumbar) 2 years ago. His medical history was unremarkable and he denied hypertension, diabetes mellitus, trauma and coagulation disorders. We performed decompression and posterolateral fusion (the fourth lumbar ~ the first sacrum). Due to previous surgery, there was dural adhesion and unintentional dural tear and CSF leakage occurred during surgery. Dural tear was sutured immediately in water-tight fashion and a drain was inserted to the subfascial space. Otherwise, surgery was uneventful and the patient was well recovered from anesthesia. After the surgery, serosanguineous fluid including cerebrospinal fluid came out through the drain. The drainage volume was gradually reduced and the drain was removed on the 8^th day after the surgery. However, serosanguineous ooze including CSF was persistent from the drain removal site until the 22^nd day. Five to ten gauzes (10 cm ×7.5 cm) were soaked everyday and the patient complained of a headache and dizziness. The exudates gradually reduced and the ooze stopped from the drain removal site on the 25^th day. The wound was healing well and headache and dizziness also improved. He was discharged from the hospital on the 30^th day. Yet, headache and dizziness came back after the discharge and he came to the emergency department presenting with fever, headache, altered level of consciousness and slurred speech on the 44^th day after the surgery. Glasgow comma scale score on the arrival was 8. A brain CT was immediately performed and revealed ventriculomegaly (Fig. 1). A brain MRI showed meningeal enhancement suggesting meningitis (Fig. 2). A lumbar MRI showed fluid collection in the lumbosacral region, the operation site (Fig. 3). Blood test results showed elevated white blood cell (WBC) (17990 /ul), elevated C-reactive protein (3.348 mg/dl). CSF test results showed elevated WBC (880 /ul), neutrophil and protein (213.2 mg/dl), reduced CSF glucose/blood glucose ratio (29/144). Based on the imaging study results and laboratory results, meningitis was suspected and the patient was admitted to the intensive care unit (ICU) then antibiotic therapy was commenced. CSF culture was requested but there was no growth. On the following day of the admission, incision & drainage was performed to drain the fluid in the lumbosacral region and a drain was inserted. Fluid culture was requested with the drainage but there was no growth. After the treatment, the level of consciousness and symptoms seemed to improve but dyspnea was developed and he became comatose on the 54^th day after the surgery (the 11^th day of re-admission). Brain CT showed cerebellar edema and hemorrhage (Fig .4). External ventricular derivation was performed as an emergency operation for decompression and he was cared in the ICU. However, his vital signs were unstable and he remained comatose and finally expired after 23 days after the operation.

Fig. 1
Computed tomography (CT) findings for the brain: Moderate ventriculomegaly.

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Fig. 2
Magnetic resonance imaging (MRI) findings for the brain (axial Gd-enhanced T1): Prominent enhancement of meninges and moderate ventriculomegaly.

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Fig. 3
MRI findings for the lumbar spine (sagittal Gd-enhanced T1): Fluid collection at the laminectomy site and around the fixators.

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Fig. 4
CT findings for the brain: Acute cerebellar hemorrhage with diffuse parenchymal swelling in both cerebellar hemispheres.

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Most of spontaneous intracranial hemorrhages before and after surgery are associated with use of anti-coagulant drugs, high arterial blood pressure, intracranial vascular malformation and neck vein obstruction due to excessive rotation. However, some reported that CSF leakage after dural tears may cause intracranial hemorrhage after spinal surgeries.4) The frequency of dural tears during spinal surgery is reported to be 3.5~17.4%.1) RCH after spinal surgery was first reported by Chadduck5) in 1981 and, in all published cases of RCH to date, RCH was associated with neurologic symptoms caused by dural tears and leakage of CSF.6)

According to the literature, RCH can occur after any types of spinal surgeries if CSF leakage can develop during surgery or after surgery regardless age, sex and the causes of diseases.6) Although the underlying pathophysiology of RCH is not yet clear, most theories seem to be related with two facts. RCH is resulted from CSF loss during and after surgery and is venous origin.7) The assumption that RCH is venous in origin is supported by the evidence that arterial hemorrhage occurs unilaterally while RCH occurs bilaterally in the cerebellar sulci and cerebellar vermis, where the cerebellar draining veins are located. The assumption that CSF leakage during surgery or after surgery causes RCH is supported by the published case reports in which CSF leakage was associated with the development of RCH.3) The development of RCH is extremely rare in patients who underwent lumbar puncture where loss of CSF is small in volume and low in speed. For the development of RCH, rapid and large amount of CSF leakage should be preceded. Some authors maintained that rapid leakage of large amount of CSF induces sagging of cerebellum.7) The cerebellar sag can cause arterial infarction or venous occlusion and lead to reperfusion hemorrhage or venous hemorrhage secondary to elevated venous pressure.7) Although most authors agree upon the view that rapid leakage of a large amount of CSF should be preceded, this is controversial because RCH has been reported after lumbar puncture in two cases8) and the volumes of CSF loss were not measured in most cases.

This complication can be immediately detected by brain CT or brain MRI. According to Brockmann,9) RCH is bilaterally seen in the upper portion of the cerebellar vermis and folia of cerebellar where venous origin of hemorrhage is seen. The bleeding is shown as clear curvy patterns including alternative high density area (blood) and low density area (cerebellum) on imaging studies and this typical pattern is described as 'zebra sign'. Since 'zebra sign' is considered to be typically seen after post-operative CSF leakage, CSF leakage should be considered when this pattern is shown on imaging studies.

RCH can be asymptomatic and be accidentally found by imaging studies. RCH can cause dizziness, headache, non-specific neurologic symptoms, cerebellar abnormality and even death.3) In general, small amount of bleeding responds well to conservative treatment with close observation by serial imaging studies. Nevertheless, large amount of hemorrhage requires immediate surgical decompression due to a high risk of acute obstructive hydrocephalus and brain stem compression.2, 6) CSF diversion is required if compression of the fourth ventricle and secondary non-communicating hydrocephalus are developed.6) In case of large amount of hemorrhage, satisfactory outcomes such as full recovery or mild neurologic symptoms are reported in more than 50% of the cases and death was reported in 10~15% of the cases.10)

Similar to previous reports, unintentional dural tears and CSF leakage also occurred in our case. Despite of the immediate suture, CSF was leaked through the drain. The patient initially complained of dizziness and mild headache but the symptoms were alleviated. However, meningitis accompanied with fever, headache, altered level of consciousness was developed and antibiotic therapy was commenced. During antibiotic therapy, RCH was developed on the 54^th day after the surgery (the 10^th day of re-admission) and external ventricular derivation was performed for decompression but he expired.

In previous reports, RCH after spinal surgery was immediate perioperative complication. But in our case, RCH was developed on the 54^th day after the surgery with history of meningitis. Cerebellar hemorrhage was reported as a complication of meningitis but was not reported as a complication of meningitis after spinal surgery. So, the cause of RCH was unclear whether CSF leakage or meningitis in our case.

The outcome of RCH after spinal surgery is generally satisfactory but we must bear in mind that RCH can be fatal in patients with altered level of consciousness, headache, dizziness and non-specific symptoms to cerebellar or cerebral lesions. Thus, special attention should be paid to prevent dural damage during spinal surgery or minimize CSF leakage when there are dural damages and tears during spinal surgery, and CT and MRI should be promptly performed for symptomatic patients.