Isolated symptomatic occlusion of the anterior cerebral artery (ACA) is a rare condition and until date, only few cases regarding the revascularization of the ACA have been reported. This paper reports on successful attempt to revascularize the ACA using superficial temporal artery (STA) in patient with isolated symptomatic occlusion of the ACA. A 69-year-old man presented with several episodes of transient weakness involving left lower extremity. Cerebral angiography showed occlusion of the right ACA at the A2 segment. After medical treatment failure, the patient underwent STA-ACA bypass surgery. Subsequent to surgery, there was immediate disappearance of transient ischemic attack and follow-up angiography showed favorable revascularization of the ACA territory. Bypass surgery can be considered in the patients with symptomatic occlusion of the ACA, who have experienced failure in medical treatment.
Anterior cerebral artery; Superficial temporal artery; Occlusion; Bypass
Coexistence of cranial and spinal subdural hematomas is rare and only a few cases have been reported in the literature. Herein, we report a case of cranial and spinal subdural hematomas after previous head trauma. As the pathogenesis of simultaneous intracranial and spinal subdural hematoma yet remains unclear, we developed an alternative theory to those proposed in the literature for their coexistence, the migration of blood through the subdural space.
Concomitant disease; Cranial subdural hematoma; Spinal subdural hematoma; Migration; Subdural space
Ossified lipoma or osteolipoma are rarely reported. It is defined as a histologic variant of lipoma that has undergone osseous metaplasia. Osteolipoma presents with a dominant osseous component within a lipoma. We report a case of a histologically confirmed osteolipoma on the nuchal ligament independent of bone. The patient was a 51-year-old female who presented with a 5-year history of a painless, progressively enlarging mass on the posterior neck. Computed tomography and magnetic resonance imaging showed a circumscribed mass compatible with fat between the C2 and C6 spinous processes with a large calcified irregular component. The mass with dual components was totally removed under general anesthesia and no recurrence was observed after 6 months of follow-up. We also reviewed the clinicopathologic features of previously reported osteolipomas in the literature and suggest that although osteolipoma is a rare variant of lipoma, it should be considered in the differential diagnosis when a lipoma of the posterior neck mixed with a bony component is encountered.
Lipoma; Osteolipoma; Ossified lipoma; Ossifying lipoma; Lipomatous tumor
Ependymoma can spread via cerebrospinal fluid, but late spinal recurrences of intracranial tumor are very rare. We describe a case of a 33-year-old male who presented with multiple, delayed, recurrent lesions in the spinal cord from an intracranial ependymoma. The patient underwent gross total resection and postoperative radiation therapy 14 years prior to visit for a low grade ependymoma in the 4th ventricle. The large thoraco-lumbar intradural-extramedullary spinal cord tumor was surgically removed and the pathologic diagnosis was an anaplastic ependymoma. An adjuvant whole-spine radiation therapy for residual spine lesions was performed. After completion of radiation therapy, a MRI showed a near complete response and the disease was stable for three years.
Fourth ventricle ependymoma; Delayed spinal recurrence; Anaplastic transformation
Upward migration of the peritoneal catheter of a subgaleo-peritoneal (SP) shunt and coiling into the subgaleal space is an extremely rare complication of a SP shunt. A 32-year-old male patient visited our hospital presenting with a large skull defect due to a prior craniectomy performed elsewhere. The patient underwent a cranioplasty with methylmetacrylate, but subsequently developed progressive pseudomeningocele and subgaleal cerebrospinal fluid (CSF) collection. The patient underwent CSF diversion via a SP shunt. After SP shunting, the pseudomeningocele disappeared completely. Six months later, the patient presented with progressive scalp swelling. Skull X-ray showed migration and coiling of the distal catheter of the SP shunt. The patient was treated by removing the entire shunt catheter and the dura was covered with a subgaleal flap. We would like to report our experience with a very rare complication of subgaleo-peritoneal shunting.
Subgaleo-peritoneal shunt; Migration; Pseudomeningocele
We report a case of infantile fibrosarcoma in an 8-month-old boy manifested as a right-sided lower leg mass. Repeated local recurrence and distant metastasis were noted during the following three-year period. Whole body fluoro-deoxyglucose positron emission tomography scan revealed an asymptomatic metastasis involving the fourth lumbar vertebrae. The patient received chemotherapy (VAC regimen) with Cyberknife® stereotactic hypofractionated radiotherapy (26 Gy; 4 fractions). This treatment reduced tumor size by 23% without acute radiation toxicity even after 33 months. This case suggests that combining chemotherapy and this form of radiotherapy may be safe and effective against childhood spinal metastasis.
Cyberknife®; Hypofractionated radiotherapy; Infantile fibrosarcoma; Spinal metastasis
Charcot spine is a progressive and destructive process that affects the vertebral bodies, intervertebral discs, and posterior facets. It is the result from repetitive microtrauma in patients who have decreased joint protective mechanisms due to loss of deep pain and proprioceptive sensation, typically because of spinal cord injury. The objective of the study is to report an unusual case of Charcot spine, as a late complication of traumatic spinal cord injury, treated by a circumferential arthrodesis performed with a single staged posterolateral costotransversectomy approach.
Charcot spine; Spinal cord injury; Circumferential arthrodesis; Posterolateral costotransversectomy approach
Charcot spinal arthropathy is a rare, progressive type of vertebral joint degeneration that occurs in the setting of any preexisting condition characterized by decreased afferent innervation to the extent that normal protective joint sensation in the vertebral column is impaired. The authors report on a case of Charcot arthropathy of the lower lumbar spine mimicking a spinal tumor following cervical cord injury.
Charcot arthropathy; Spinal cord injury; Sacrum
The glomus tumor of the peripheral nerve is one of the mesenchymal tumors originating in the epineurium, and is extremely rare. A 56-year-old man presented complaining of lancinating pain on the left thigh, which was provoked by pressure or exercise. Subsequent image study revealed a mass in the femoral nerve. Total surgical excision with the aid of intraoperative ultrasonography was performed and the pain was successfully controlled. The authors report an unusual case of a patient diagnosed with glomus tumor in peripheral nerve, with a review of the clinical features, imaging, and pathological findings.
Glomus tumor; Peripheral nerve; Femoral nerve
The authors present a rare case of adult moyamoya disease in which a patient experienced rapid progression of cerebral infarction after intraventricular hemorrhage (IVH). A healthy 39-year-old woman was admitted to our hospital with sudden headache, a decreased level of consciousness and mild tetraparesis. Initial magnetic resonance imaging revealed small cerebral infarction and IVH. Although the patient underwent conservative therapy including hypervolemia, hemodilution, keeping moderate hypertension and administration of a free radical scavenger, she showed a fulminant clinical course of cerebral infarction. The authors discuss the possible pathophysiology and suggest the treatment for such cases.
Moyamoya disease; Cerebral infarction; Intraventricular hemorrhage
Hemangioblastomas are sporadic tumors found in the cerebellum or spinal cord. Supratentorial hemangioblastomas are rare, and those with meningeal involvement are extremely rare and have been reported in only approximately 130 patients. Here, we report the case of a 51-year-old female patient with supratentorial meningeal hemangioblastoma detected 5 years after surgical resection of an infratentorial hemangioblastoma associated with von Hippel-Lindau disease. Patients with von Hippel-Lindau syndrome are at risk for developing multiple hemangioblastomas, with new tumor formation and growth and possible meningeal infiltration. Regular lifelong follow-up in at-risk patients is recommended and should include the differential diagnosis of dural-based tumors such as angioblastic meningioma and metastatic renal cell carcinoma.
Angioblastic meningioma; Hemangioblastoma; Supratentorial; Von Hippel-Lindau disease
Lymphomatosis cerebri is considered a diffuse form of primary central nervous system lymphoma and very rare. It is not well recognized and may be misdiagnosed with infiltrating tumors, degenerative disorders, ischemic diseases, and infectious diseases developed in the brain. Awareness of the possibility of this rare disease and early biopsy are required for differential diagnosis and preventing poor clinical outcomes. We report a case with lymphomatosis cerebri who presented with rapid neurological deteriorations and review the relevant literatures.
Lymphomatosis cerebri; Primary central nervous system lymphoma; Biopsy
We present a case report to remind surgeons of this unusual complication that can occur in any surgery, even posterior cervical spine surgery under general anesthesia and discuss its causes, treatment methods, and the follow-up results in the literature. The peripheral Tapia's syndrome is a rare complication of anesthetic airway management. Main symptoms are hoarseness of voice and difficulty of tongue movement. Tapia's syndrome after endotracheal general anesthesia is believed to be due to pressure neuropathy of the vagus nerve and the hypoglossal nerve caused by the endotracheal tube. To our knowledge, no report has been published or given an explanation for Tapia's syndrome after posterior cervical spine surgery. Two patients who underwent posterior cervical surgery complained hoarseness and tongue palsy postoperatively. There is no direct anatomical relation between the operation, the vagus nerves and the hypoglossal nerves, and there is no record of displacement or malposition of the endotracheal tube. After several months, all symptoms are resolved. To avoid this problem in posterior cervical spine surgery, we suggest paying special attention to the position of the endotracheal tube to avoid excessive neck flexion before and during the positioning of the patient.
Tapia's syndrome; Posterior cervical spine surgery; Hypoglossal nerve; Vagus nerve
The prognosis of solitary plasmacytoma varies greatly, with some patients recovering after surgical removal or local fractional radiation therapy, and others progressing to multiple myeloma years later. Primary detection of progression to multiple myeloma is important in the treatment of solitary plasmacytoma. There have been several analyses of the risk factors involved in the early progression to multiple myeloma. We describe one case of solitary plasmacytoma of the lumbar vertebra that was treated with surgical decompression with stabilization and additional radiotherapy. The patient had no factors associated with rapid progression to multiple myeloma such as age, size, immunologic results, pathological findings, and serum free light chain ratio at the time of diagnosis. However, his condition progressed to multiple myeloma less than two months after the initial diagnosis of solitary plasmacytoma. We suggest that surgeons should be vigilant in watching for rapid progression to multiple myeloma even in case that the patient with solitary plasmacytoma has no risk factors for rapid progression to multiple myeloma.
Solitary plasmacytoma; Serum free light chain; Multiple myeloma; Lumbar vertebrae
Acute subdural hematoma is usually a neurological emergency that requires hematoma evacuation or close observation. However, spontaneous resolutions of an acute subdural hematoma without surgical interventions have been reported rarely. We report on a case who showed rapid resolution of an acute subdural hematoma with neurological improvement and review the relevant literatures.
Rapid; Resolution; Subdural hemorrhage
Isolated oculomotor nerve palsy (ONP) attributable to mild closed head trauma is a distinct rarity. Its diagnosis places high demands on the radiologist and the clinician. The authors describe this condition in a 36-year-old woman who slipped while walking and struck her face. Initial computed tomography did not reveal any causative cerebral and vascular lesions or orbital and cranial fractures. Enhancement and swelling of the cisternal segment of the oculomotor nerve was seen during the subacute phase on thin-sectioned contrast-enhanced magnetic resonance images. The current case received corticosteroid therapy, and then recovered fully in 13 months after injury. Possible mechanism of ONP from minor head injury is proposed and previous reports in the literature are reviewed.
Computed tomography; Head injury; Magnetic resonance imaging; Oculomotor nerve; Oculomotor nerve palsy
The cerebrospinal fluid tap test (CSFTT) is recommended as a key step in the diagnosis of idiopathic normal pressure hydrocephalus (iNPH). While there is no generally accepted evaluation period for ascertaining a CSFTT responder, a substantial number of patients are evaluated only once within 24 hours of the test for improvement in gait. We report an iNPH patient with a favorable response to shunt surgery, who was first judged a non-responder by this standard, though subsequently was judged a responder in virtue of repetitively testing gait over 7 days. A 68-year-old man presented with progressive impairment of gait, balance, and memory. He was diagnosed as iNPH with an Evans' ratio of 0.35. At first hospitalization, change in gait was evaluated 24 hours after the CSFTT. He didn't show any significant improvement and was judged as a non-responder. However, at the second CSFTT, we repetitively tested his change in gait over seven days. Forty-eight hours after the tap, he showed significant improvement in his gait. He was then confirmed as a responder. After the operation, the gait difficulties were almost fully resolved. Further studies developing the standard procedure of the CSFTT should be considered.
Cerebrospinal fluid shunts; Normal pressure hydrocephalus; Predictive value of tests
Liver abscess following ventriculoperitoneal (VP) shunting occurs very rarely. We report an unusual case of multiple liver abscesses caused by Staphylococcus capitis in a 50-year-old compromised woman due to a complicating VP shunt infection. We reviewed the nine cases of VP shunt complications reported in the English literature, and speculated that the most likely pathogenetic mechanism in our case is an infected peritoneal tip that migrated to and penetrated the liver, which subsequently caused the formation of multiple liver abscesses. The patient was successfully treated with percutaneous aspiration, drainage of the abscesses, intravenous antibiotics, and shunt revision. Awareness and vigilance of the possibility of liver abscess formation caused by VP shunt infection will help establish an early accurate diagnosis and therapeutic strategy.
Liver abscess; Catheter-related infections
Spinal extradural arachnoid cyst (SEAC) is a rare disease and uncommon cause of compressive myelopathy. The etiology remains still unclear. We experienced 2 cases of SEACs and reviewed the cases and previous literatures. A 59-year-old man complained of both leg radiating pain and paresthesia for 4 years. His MRI showed an extradural cyst from T12 to L3 and we performed cyst fenestration and repaired the dural defect with tailored laminectomy. Another 51-year-old female patient visited our clinical with left buttock pain and paresthesia for 3 years. A large extradural cyst was found at T1-L2 level on MRI and a communication between the cyst and subarachnoid space was illustrated by CT-myelography. We performed cyst fenestration with primary repair of dural defect. Both patients' symptoms gradually subsided and follow up images taken 1-2 months postoperatively showed nearly disappeared cysts. There has been no documented recurrence in these two cases so far. Tailored laminotomy with cyst fenestration can be a safe and effective alternative choice in treating SEACs compared to traditional complete resection of cyst wall with multi-level laminectomy.
Arachnoid cyst; Cerebrospinal fluid; Thoracolumbar spine
Intracerebral schwannomas are rare and there have been none reported in Korea. We present the case of a 25-year-old man with newly developed right-side weakness and recent seizure aggravation. His seizures started approximately 9 years prior to admission. At that time, a 1 cm diameter intra-axial enhancing mass at the left precentral gyrus was found on magnetic resonance image (MRI). After 9 years of observation and treatment with antiepileptic medication, an MRI taken due to symptom aggravation revealed peri-tumoral cyst formation with tumor enlargement. The tumor was surgically removed. Subsequently, right-side weakness diminished and there was good seizure control. Pathologic diagnosis was schwannoma. Schwannoma is a very rare tumor and there are no pathognomonic findings on radiologic images; thus, it is challenging to make a correct diagnosis. However, considering the natural course and excellent prognosis after surgical treatment of this kind of intra-axial mass with benign features, early surgery for diagnosis and proper treatment is highly recommended.
Intracerebral schwannoma; Supratentorial; Precentral gyrus; Treatment; Seizure
A 55-year-old female patient presented with lower back pain and neurogenic intermittent claudication and underwent L3-L4 posterolateral fusion. To prepare the bone fusion bed, the transverse process of L3 and L4 was decorticated with a drill. On the 9th post-operative day, the patient complained of a sudden onset of severe abdominal pain and distension. Abdominal computed tomography revealed retroperitoneal hematoma in the right psoas muscle and iatrogenic right L3 transverse process fracture. Lumbar spinal angiography showed the delayed hematoma due to rupture of the 2nd lumbar artery pseudoaneurysm and coil embolization was done at the ruptured lumbar artery pseudoaneusyrm. Since then, the patient's postoperative progress proceeded normally with recovery of the hemodynamic parameters.
Lumbar artery; Spinal fusion; Posterolateral fusion
We report a very rare case of cervical compressive myelopathy by an anomalous bilateral vertebral artery (VA) entering the spinal canal at the C1 level and compressing the spinal cord. A 70-year-old woman had been suffering from progressive gait disturbance. Magnetic resonance imaging revealed that a bilateral VA at the V4 segment had abnormal courses and caused compression to the high cervical cord. VA repositioning was performed by anchoring a suture between the artery and around the arachnoid membrane and dentate ligament, and then, microvascular decompression using a Teflon sponge was done between the VA and the spinal cord. The weakness in the patient improved in the lower extremity after the operation. Anomalous VA could be one of the rare causes of cervical compressive myelopathy. Additionally, an anchoring suture and microvascular decompression around the VA could be a sufficient and safe method to indirectly decompress the spinal canal.
Vertebral artery anomaly; Cervical myelopathy; Microvascular decompression
To present a rare case of a cystic giant schwannoma of the sacrum mimicking aneurysmal bone cyst (ABC). A 54-year-old man visited our institute complaining left leg weakness and sensory change for several years. Magnetic resonance imaging revealed a large multilocular cystic mass with canal invasion and bone erosion confined to left S1 body. The lesion showed multiple septal enhancement without definite solid component. Initially the tumor was considered as ABC. The patient underwent grossly-total tumor resection with lumbosacral reconstruction via posterior approach. The tumor was proved to be a cystic schwannoma. The postoperative course was uneventful and the patient was relieved from preoperative symptoms. We present a rare case of pure cystic giant schwannoma confined to sacrum mimicking ABC. The surgical treatment is challenging due to the complex anatomy of the sacrum. Schwannoma should be considered in the differential diagnosis of osteolytic sacral cysts.
Giant schwannoma; Sacrum; Cystic degeneration; Aneurysmal bone cyst
Chronic hypoventilation due to injury to the brain stem respiratory center or high cervical cord (above the C3 level) can result in dependence to prolonged mechanical ventilation with tracheostomy, frequent nosocomial pneumonia, and prolonged hospitalization. Diaphragm pacing through electrical stimulation of the phrenic nerve is an established treatment for central hypoventilation syndrome. We performed chronic phrenic nerve stimulation for diaphragm pacing with the spinal cord stimulator for pain control in a quadriplegic patient with central apnea due to complete spinal cord injury at the level of C2 from cervical epidural hematoma. After diaphragmatic pacing, the patient who was completely dependent on the mechanical ventilator could ambulate up to three hours every day without aid of mechanical ventilation during the 12 months of follow-up. Diaphragm pacing through unilateral phrenic nerve stimulation with spinal cord stimulator was feasible in an apneic patient with complete quadriplegia who was completely dependent on mechanical ventilation. Diaphragm pacing with the spinal cord stimulator is feasible and effective for the treatment of the central hypoventilation syndrome.
Apnea; Central hypoventilation; Diaphragm pacing; Spinal cord stimulator; Spinal cord injury
Split cord malformations (SCMs) usually present in childhood, and are rarely reported in adults. And also, a cervicothoracic SCM associated with tethered cord syndrome has very rarely been reported in the literature. We report a case of SCM associated with tethered cord and spina bifida in an adult. This report describes the case of a 34-year-old woman who presented for evaluation of neck pain, back pain, and intermittent paraparesis of several months duration. The MRI and CT showed a SCM at the cervicothoracic level and a fibrous septum at the thoracic level. She underwent surgery for the SCM and tethered cord syndrome, and was followed for 7 years. Patient presented complete recovery in the follow-up. The authors discuss this unusual lesion and describe the anatomical relationship of the level of cord duplication and fibrous septum.
Split cord malformation; Adult; Tethered cord syndrome