Effectiveness of intraarterial administration of fasudil hydrochloride for preventing symptomatic vasospasm after subarachnoid hemorrhage.

OBJECTIVE: We examined the effect of intraarterial administration of fasudil hydrochloride (IAFC), a Rho kinase inhibitor, for the prevention of symptomatic vasospasm after SAH by evaluating cerebral circulation. METHODS: We evaluated IAFC cases of 57 sides of 38 patients (12 men and 26 women, average age 60.2 years old) diagnosed with aneurysmal subarachnoid hemorrhage (SAH) from February 2012 to November 2012. All cases were treated by clipping or coil embolization within 48 h after onset. Indication for IAFC was the existence of a spastic change on follow-up digital subtraction angiography (DSA) compared with that of onset. RESULTS: Clipping was performed in 30 cases and coil embolization in 8 cases. IAFC was performed an average of 6.6 days after onset. Color gradient mapping demonstrated reduction of the circulation time after IAFC compared with before IAFC on 39 sides, no change on 15 sides, and extension on 3 sides. Average arterial circulation time before IAFC was 2.25 ± 0.57 s and after IAFC was 1.95 ± 0.55 s. IAFC significantly shortened average arterial circulation (P = 0.005). No case developed symptomatic vasospasm after IAFC. CONCLUSION: IAFC significantly reduced the cerebral circulation time after aneurysmal SAH and might be effective for the prevention of symptomatic vasospasm.

[Analysis of Rupture during Follow-up of Unruptured Aneurysm].

OBJECT: To elucidate clinical aspects of ruptured aneurysms, we retrospectively investigated associations between risk factors and ruptured and unruptured cases during conservative management. METHODS: Two hundred and twenty-nine patients with 291 unruptured cerebral aneurysms treated between 2000 and 2012 were analyzed. Mean duration of observation was 62 months (1183.4 person-years). We investigated the following six risk factors: history of subarachnoid hemorrhage;multiplicity;location of aneurysms;aneurysm size ≤5mm;bleb or irregular forms;and follow-up period <1 year. RESULTS: Twenty-two aneurysms in 22 patients (19 women;86.4%) ruptured during this study. The annual rate of rupture was 1.86%. In ruptured cases, mean age was 66.7 years. According to univariate analysis, aneurysm size≥5mm(p=0.000), bleb or irregular form(p=0.006)and duration of observation<1 year (p =0.000) were significantly associated with aneurysmal rupture. In multivariate analysis of these factors, aneurysm size≥5mm(p =0.0188;odds ratio(OR), 3.4;95% confidence interval (CI), 1.2-9.7) and duration of observation<1 year (p=0.006;OR, 5.0;95% CI, 1.6-14.9) represented independent risk factors for aneurysm rupture. CONCLUSIONS: The results of this study were almost the same as those of the UCAS Japan study. In addition, duration of observation <1 year was a risk factor for aneurysm rupture. When we decide on surgical treatment after considering factors such as aneurysm size, form, and surgical risk, surgery should be performed as soon as possible.

[An operated case of a giant fusiform aneurysm of the middle cerebral artery with alternating radiological findings within a short period].

We report an operated case of a giant fusiform aneurysm of the middle cerebral artery (MCA). An 18-year-old man presenting with a 6-month history of severe left temporal headache was admitted to our department. On admission, MRI revealed a mass lesion measuring 45 mm in diameter in the left frontal lobe. A left carotid angiogram revealed aneurysmal dilatation and stenosis in the M2 portion of the left MCA, which was diagnosed as a giant fusiform dissecting aneurysm. An intracarotid amobarbital test (Wada test)demonstrated ischemic tolerance to occlusion of the parent artery. Spontaneous occlusion of the parent artery and obliteration of the aneurysmal lesion incidentally occurred 15 days after admission. Follow-up 3D-CT angiography revealed recurrence of the aneurysmal dilatation in the same segment of the artery 6 days after the spontaneous obliteration. The lesion was then successfully resected without revascularization. Histopathological examination revealed a pseudolumen and loss of the three-layer structure of the aneurysmal wall. The postoperative course was uneventful and the patient was discharged without neurological deficits. We present the case report and a review of the literature.

Symptomatic vasospasm in elderly patients with aneurysmal subarachnoid hemorrhage: comparison with nonelderly patients.

OBJECTIVE: To clarify the influence of age on the occurrence of symptomatic vasospasm (SVS), we retrospectively compared 34 elderly (over 70 years) and 71 nonelderly patients with aneurysmal subarachnoid hemorrhage (SAH). METHODS: Between 2008 and 2010, at our hospital 105 patients (Hunt and Kosnik grades I-IV) underwent aneurysm surgery within 72 h of the insult. They were divided into four groups based on their age (younger/older than 70 years) and treatment (aneurysmal clipping or coiling). In all patients, we used the same protocol, which included the delivery of intrathecal urokinase and intravenous fasudil chloride; in patients with angiographic evidence of vasospasm, we also injected fasudil chloride intra-arterially. RESULTS: Among the elderly patients, 4.3% of those treated by clipping and 9.1% of those treated by coiling experienced SVS; the comparative incidence in younger patients was 6.5% and 4.0%, respectively. The differences were not statistically significant (p = 0.40). The ratio of ventriculo peritoneal (VP) shunts was higher in the elderly patients (p = 0.00007). The incidence of favorable treatment outcomes was significantly lower in elderly patients (p = 0.00004). CONCLUSION: Under our treatment protocol, patient age did not affect the incidence of SVS. Our protocol may be effective for the prevention of SVS after aneurysmal SAH regardless of patient age.

Usefulness of three-dimensional computed tomography to quantify the subarachnoid hemorrhage volume: prediction of symptomatic vasospasm.

We quantified the subarachnoid hemorrhage (SAH) volume in 64 patients on three-dimensional computed tomography (3D-CT) scans and studied the correlation between the SAH volume and the occurrence of symptomatic vasospasm (SVS). We studied 64 patients with SAH onset (day 0) and on days 1, 4, 7, and 14. We compared the hematoma volume by 3D-CT with 2D-CT on day 0 and examined the correlation between the hematoma volume and the occurrence of SVS. The hematoma volume, including the volume of normal structures, was automatically calculated (V1). The volume of normal structures manifesting identical CT numbers was previously calculated in patients without intracranial lesions (V2). The total hematoma volume was defined as V1 minus mean value of V2 (= 12 ml). The mean hematoma volume by 3D-CT was 48 ± 12 ml and by 2D-CT was 31 ± 45 ml (mean ± SD, n = 64). The hematoma volume was significantly larger by 3D-CT than by 2D-CT (p < 0.05). At all time points, the hematoma volumes were significantly larger in patients with than without SVS. We developed a new method for the quantitative determination of the SAH volume by 3D-CT. This method may allow us to quantify the volume of SAH in clinical studies of cerebral vasospasm.

[An operated case of a meningioma causing acute subdural hematoma].

We report a rare case of a meningioma causing acute hematoma. A 67-year-old woman presented with sudden headache. No evidence of trauma was seen. CT demonstrated a subdural hematoma in the convexity of the fronto-temporal lobe. Magnetic resonance imaging showed marked signal heterogeneity in the convexity of the frontal lobe. One week later, the patient underwent hematoma evacuation and tumor resection including the attached dura mater. The histological diagnosis was meningothelial meningioma. The clot was connected directly to the tumor and the origin of the subdural hematoma was identified as the meningioma. Postoperative course was uneventful, and the headache improved. Meningiomas have a relatively benign course but rarely present with hemorrhage. Surgical exploration is the effective and recommended treatment.

Fatal subarachnoid hemorrhage caused by Aspergillus arteritis without angiographic abnormalities.

No source of bleeding is detected by angiogram in 15-20% of patients with nonaneurysmal subarachnoid hemorrhage (SAH). This negative angiographic finding might suggest a benign prognosis. We describe a case of fatal SAH caused by Aspergillus arteritis without formation of fusiform dilatation or aneurysms. A 76-year-old man with a 2-month history of progressive visual loss due to pachymeningitis around the optic nerves suffered from SAH in the bilateral sylvian fissures. Repetitive serum galactomannan assay and angiography showed no abnormality. Post mortem examination revealed marked proliferation of Aspergillus in the granulomas of the frontal base dura mater. In addition, major trunks and several branches of the bilateral middle cerebral arteries were invaded by Aspergillus hyphae, which destroyed the walls in the absence of dilatation and aneurysms. Invasive aspergillosis of the CNS often forms a mycotic aneurysm. However, four autopsy cases of nonaneurysmal SAH due to invasive aspergillosis have been reported. The present case is the second autopsy case of Aspergillus arteritis without angiographic abnormality, resulting in fatal SAH. Aggressive and continuous antifungal therapy is absolutely necessary in suspected cases of invasive aspergillosis of the CNS, even if angiography is negative and therapeutic markers of aspergillosis are normal.

[Operated case of a ruptured aneurysm arising from fenestration of the vertebral artery].

Aneurysm arising from duplication of the vertebral artery (VA) is rare. We report a surgical case of an aneurysm of VA fenestration and provide a literature review. A 42-year-old man suffered from sudden onset of severe headache. CT image revealed subarachnoid hemorrhage predominantly in the posterior fossa. Digital subtraction angiography showed a fenestration of the right VA and the aneurysm arising from the proximal limb of the fenestration. Neck clipping of the aneurysm was performed because embolization was difficult due to the wide neck. The patient was discharged without deficits. Seven cases of an aneurysm of the VA fenestration have been reported. Five cases underwent coil embolization, and 2 underwent craniotomy, trapping, and resection. This is the first case of neck clipping of an aneurysm of the VA fenestration.

[Childhood transverse sinus dural arteriovenous fistula treated with endovascular and direct surgery: a case report].

Infantile dural arteriovenous fistula is a rare cerebrovascular malformation carrying a poor prognosis with an anatomic cure of only 9%. Endovascular embolization is mainly selected to treat this entity, aiming to obtain normal development of the patients. We present a case of a 20-month-old girl with epilepsy. Digital subtraction angiography revealed a dural arteriovenous fistula involving the right transverse sinus. The arteriovenous fistula was fed by multiple dural branches from the middle meningeal, occipital, meningohypophyseal, and anteroinferior cerebellar arteries. The right transverse sinus was transvenously embolized with platinum coils. Although the shunt flow remained, the patient was liberated from epilepsy. Nine months later, the patient suffered from a recurrence of epilepsy. Digital subtraction angiography demonstrated some increase in shunt flow. Right middle meningeal, occipital, posterior deep temporal, and tentorial arteries were transarterially embolized using N-butyl cyanoacrylate, followed by complete surgical resection of the right transverse sinus. The shunt flow disappeared after surgery, and her epilepsy improved significantly. Our experience suggests that the combination of endovascular and surgical treatment is effective for recurrent infantile dural arteriovenous fistula.

[Flow dynamics analysis in patients with a ruptured middle cerebral artery aneurysm. A case report].

Despite advancement of diagnostic and treatment modalities, subarachnoid hemorrhage (SAH) is still an entity of neurosurgical emergency with poor outcome. Recent reports indicated that hemodynamic stress might play an important role in rupture or the growth of cerebral aneurysms, but there is no consensus about how or which hemodynamic factor contribute to this phenomenon. In this report, magnetic resonance (MR)-based flow dynamics analysis was performed for a patient with SAH and the data obtained were directly compared with intraoperative findings. This 74-year-old woman was admitted for sudden onset headache. Head computed tomography scan showed SAH on the right sylvian fissure and intracerebral hematoma on the right temporal tip. Digital subtraction angiography showed a right middle cerebral artery aneurysm, which was considered to be the ruptured one. The aneurysm had two blebs, and the bleb around the aneurysm tip was exposed to low magnitude and high oscillation of wall shear stress (WSS). On the other hand, another bleb was exposed to high magnitude and low oscillation of WSS. Next day, the patient underwent open surgery and intraoperative findings showed the aneurysm tip was the ruptured point. MR-based flow dynamics analysis might be a useful diagnostic modality for patients with SAH. Although low magnitude and high oscillation of WSS might contribute to the aneurysm rupture, further case accumulation is necessary to reach a conclusion whether or not this is so.

[A case of symptomatic arachnoid cyst, that had been growing for 14 years, at the posterior part of the left temporal lobe].

The authors report a 54-year-old woman with headache in whom a cyst was detected at the posterior part of the left temporal lobe following a traffic accident in 1993. Symptoms had recently worsened and computed tomography demonstrated an enlarged cyst in 2007. Thereafter, the patient underwent cyst fenestration. Intraoperative findings showed multilocular cysts with calcification. Symptoms improved immediately. Histological findings showed that the cyst was composed of collagenous membrane and a monolayer of cells compatible with arachnoid cyst. These findings also showed calcification and we considered that growth of the arachnoid cyst had been induced by trauma. We expected an arachnoid cyst with a single cavity, but this lesion was multilocular. Retrospectively, cine magnetic resonance image (MRI) seemed to show multilocular cysts. Cine MRI might be an effective tool to determine whether a cyst is multilocular.

[A case of posttraumatic forehead cavernous angioma].

Cavernous angiomas of the bone are rare tumors. Skull cavernomas are even less frequent. Most cavernous angiomas of the bone are congenital tumors. In a review of the literature, we found only one case report of de novo generation of a skull cavernous angioma. We present a case of a 25-year-old woman who had experienced a head injury, and 7 years later exhibited a skull tumor at the exact region of the injury. We performed tumor resection and cranioplasty. Follow-up examinations revealed no recurrence or neurological defects. Pathological findings showed a cavernous angioma-like lesion with some atypical details. We finally diagnosed the lesion as a de novo cavernous angioma. Our case suggests that fine injury may result in de novo generation of bone cavernomas.

[Choice of intentional partial coiling for a ruptured intracranial aneurysm in the acute stage followed by clipping in the chronic stage].

The inpact of the International Subarachnoid Aneurysm Trial (ISAT) trial publication in 2002 and major technical advances in neuroimaging, endovascular devices and techniques have resulted in increasing numbers of patients with ruptured aneurysms undergoing endovascular coiling, as first-line treatment for aneurysm occlusion. We treated six cases of ruptured aneurysms with this strategy, with choice of clipping as as additional treatment following intentional partial coiling. In this series, patients ranged in age from 36 to 74 years and included one man and five women. Three aneurysms were located in AcomA, 1 in ACA, and 2 in MCA. The reasons for the choice of this strategy were clinical and angiographical spasm in 2, high general surgical risk in 2, and other reasons in 2 cases, respectively. The mean interval between the first partial coiling and final clipping was 33 days. All cases were successfully clipped without difficulties and coil removal were performed in 3 cases for follow up examination. It is proposed that the choice of this strategy contributes to progresses in overall outcomes in cases of aneurysmal subarachnoid hemorrhage.

[A successful surgical case of a hypothalamic hamartoma with gelastic seizure: a case report].

Gelastic seizure is a rare form of epilepsy defined as automatic bouts of laughter without mirth commonly associated with a hypothalamic hamartoma. Surgical treatment of hypothalamic hamartomas is associated with a high risk of complications because of the close vicinity of adjacent structures such as the optic tracts and mammillary bodies. This case was an 11-year-old girl who presented with gelastic seizure and complicated partial seizure. She developed gelastic seizure at a frequency of 10 bouts per day. She was found to have an elliptical mass close to the left hypothalamus. The signal intensity on magnetic resonance imaging (MRI) was consistent with hypothalamic hamartoma. The patient underwent surgical resection by a subtemporal approach. Pathological findings confirmed the diagnosis of hypothalamic hamartoma. Postoperative MRI demonstrated that the hypothalamic hamartoma was successfully resected. Twenty four months after surgery, complicated partial seizure in this patient has improved to Engel's class 1a and gelastic seizure has improved to Engel's class IIIa.

Cranial hemophilic pseudotumor associated with factor IX deficiency: case report.

BACKGROUND: Hemophilic pseudotumor is an unusual complication occurring in only 1% to 2% of patients with severe factor VIII or IX deficiency, and manifests as a progressive enlargement of hematoma by recurrent hemorrhage, often resulting in bone destruction or resorption due to the chronic pressure of osseous hemorrhage. Cranial hemophilic pseudotumors are extremely rare, with only 4 previous cases associated with mild or moderate factor XIII deficiency. CASE DESCRIPTION: A 24-year-old man with moderate deficiency of factor IX developed a cranial pseudotumor as a swelling in the frontal scalp. Blood coagulation profile revealed extended activated partial thromboplastin time (58.2 seconds). Factor IX analysis showed 3% of normal activity. Computed tomography and magnetic resonance imaging demonstrated an extra-axial lesion with bone destruction, enhanced rim, and signal changes consistent with chronic hemorrhage. Surgical removal was performed. Histologic examination disclosed old blood coagulum. CONCLUSIONS: This case of cranial hemophilic pseudotumor in a patient with moderate factor IX deficiency suggests that cranial pseudotumor should be considered in the differential diagnosis of cranial lesion in a patient with hemophilia, and adequate replacement therapy in the perioperative period is essential to achieve safe surgical removal.

Intracranial peripheral-type primitive neuroectodermal tumor.

A 15-year-old man presented with headache. Magnetic resonance (MR) imaging revealed a large extraaxial tumor with cyst at the right frontotemporal region. The solid part of the tumor was homogeneously enhanced on T(1)-weighted MR imaging after injection of gadolinium. Digital subtraction angiography of the external carotid artery revealed sunburst appearance corresponding to the tumor, which was fed by the right middle meningeal artery. His headache worsened and computed tomography revealed enlargement of the tumor and intracystic hemorrhage, so emergent operation was performed. At surgery, the tumor strongly adhered to the dural membrane, and was obviously extraaxial. The tumor and cyst were gross totally removed. The attachment site at the dura mater was resected. Histological examination showed solid growth of small round cells with uniform round nuclei and minimal cytoplasm. Immunohistochemical staining showed the cells were positive for MIC-2 (CD99). The MIB-1 labeling index was 53%. The histological diagnosis was peripheral-type primitive neuroectodermal tumor (pPNET). Following surgery, radiation therapy and chemotherapy were given. Ewing's sarcoma and pPNET form a family of small round cell tumors arising in the bone or soft tissue. MIC-2 is a useful marker in the differential diagnosis. Good prognosis may be attained if complete surgical excision of intracranial pPNET is achieved.

Ectopic recurrence of dysplastic gangliocytoma of the cerebellum (Lhermitte-Duclos disease): a case report.

Lhermitte-Duclos disease is a rare clinical entity characterized by slow deformation of the cerebellar lesion. A 53-year-old woman presented with Lherimitte-Duclos disease manifesting as ataxic gait, occipital headache, and loss of consciousness. Magnetic resonance imaging demonstrated striated and laminar pattern lesions in the right cerebellar hemisphere and vermis. She underwent subtotal removal of the vermis compressing the brainstem, but the cerebellar hemisphere appeared normal and was preserved. Histological findings were consistent with Lherimitte-Duclos disease. Two years later, magnetic resonance imaging revealed enlargement of the right hemispheric lesion. Intraoperative appearance of the hemisphere demonstrated marked enlargement of the folia. Subtotal removal was performed. Histological examination showed no malignant findings. Lhermitte-Duclos disease may recur, and should be carefully followed up as a low-grade neoplasm, not as a hamartomatous disorder.

Impact of anomalous origin of the ophthalmic artery from the middle meningeal artery on selection of surgical approach to skull base meningioma.

BACKGROUND: Ectopic sites of origin of the ophthalmic artery from the MMA are associated with visual complications of surgery directed along the sphenoidal wing or embolization of the MMA. CASE DESCRIPTION: Three cases of skull base meningioma in which retinal blood supply originated from the MMA are presented. In one case, the patient experienced transient blindness during selective angiography of the external carotid artery. Because surgical obliteration of the MMA might have caused visual impairment due to sacrifice of the collateral branches from the MMA to the retina during a skull base approach, we selected an approach not involving the MMA. CONCLUSIONS: In a case in which retinal blush is observed from the MMA on selective angiography of the ECA, we should select an appropriate surgical approach to avoid visual complications in cranial base surgery, taking into account a retinal blood supply.

Postpartum dissecting aneurysm of the posterior cerebral artery.

Intracranial dissecting aneurysm may cause subarachnoid hemorrhage (SAH) or infarction, and postpartum dissecting aneurysm is rare. A 30-year-old 6 days postpartum woman presented with posterior cerebral artery (PCA) dissection evolving dramatically over a short period. She had been well until 6 days after delivery when she suffered sudden onset of headache, vomiting, and unconsciousness. CT scan demonstrated SAH and digital subtraction angiography (DSA) revealed a fusiform dilatation of the left PCA (P3/P4 segment). The initial diagnosis was ruptured dissecting aneurysm, and conservative management was recommended in the acute period. DSA showed smoothening of the vascular wall 6 days after onset, and obliteration of the left P3/P4 segment was observed 13 days after onset. She was discharged without neurological deficits 26 days after onset. Postpartum SAH due to dissecting aneurysm of the PCA is rare, but should be considered in the differential diagnosis of postpartum headache.