A Case of Wallenberg's Syndrome Presenting with Spontaneous Thrombosis of a Vertebral Artery Aneurysm.

Complete spontaneous thrombosis of intracranial aneurysms is uncommon. Although this type of thrombosis is largely asymptomatic, in rare cases it can be accompanied by parent artery occlusion and ischemic stroke. There are limited reports of complete thrombosis of an unruptured aneurysm of the internal carotid artery and middle cerebral artery. Furthermore, there are no reports of occlusion of the vertebral artery caused by thrombosis of an aneurysm. The mechanisms of spontaneous thrombosis are not established. However, aneurysm morphology, arteriosclerosis, and stagnation of aneurysm flow have been suggested. Herein, we present a novel case of Wallenberg's syndrome caused by a fusiform aneurysm in which complete thrombosis of the proximal vertebral artery occurred. We discuss the mechanisms of thrombosis caused by an unruptured aneurysm, which may be useful for managing such patients who present with transient ischemic attacks.

Severe Reversible Cerebral Vasoconstriction Syndrome with Large Posterior Cerebral Infarction.

Reversible cerebral vasoconstriction syndrome is characterized by thunderclap headache and multifocal cerebral vasoconstriction. Cerebral vasoconstriction is reversible, and most cases have good prognosis. However, clinical outcome is possibly severe when it is complicated by stroke, yet detailed reports on such a case are few. We experienced a case of severe reversible cerebral vasoconstriction syndrome in a 32-year-old woman with medical history of preeclampsia 3years prior. She presented with sudden sharp headache followed by altered mental status and vasoconstriction of the bilateral posterior cerebral arteries. She was treated with intravenous and oral calcium channel blockers, edaravone, and glycerol. However, the cerebral infarction in the posterior circulation subsequently remained, and her impaired consciousness did not recover. Furthermore, although imaging findings of vasoconstriction showed improvement a day after the occurrence of symptom, the same vessels showed poor visualization 7 weeks later, which indicated the recurrence of vasoconstriction, without additional symptom due to the fixed infarction. Although most cases of reversible cerebral vasoconstriction syndrome show good prognosis, neurologists must monitor the possibility of worse clinical course and permanent neurological deficit when associated with stroke, such as cerebral infarction. Strict management and treatment are needed in these cases.

Paroxysmal Sympathetic Hyperactivity after Surgery for Cerebral Hemorrhagic Arteriovenous Malformation: A Case Report.

Paroxysmal sympathetic hyperactivity is a condition involving a sudden increase in body temperature, heart rate, blood pressure, respiratory rate, sweating, and posturing followed by severe brain injury. Most of the reported preceding disorders involve head trauma, followed by anoxic brain injury, and stroke. Here, we report an extremely rare case of 17-year-old man diagnosed with hemorrhagic arteriovenous malformation, underwent emergent surgery, was on prolonged sedation due to postoperative complications, and subsequently developed paroxysmal sympathetic hyperactivity. We recommend monitoring for paroxysmal sympathetic hyperactivity occurrence with severe brain injury patients, even when sedating.

Late-Onset Massive Epistaxis due to a Ruptured Traumatic Internal Carotid Artery Aneurysm: A Case Report.

A traumatic internal carotid artery (ICA) aneurysm is rare and difficult to treat. Trapping of ICA is commonly performed owing to the difficulty of directly approaching ICA aneurysms. Recently, coiling the aneurysm itself was recommended if possible. However, it is controversial which of methods are best to completely treat aneurysm. We present the case of a 74-year-old man, who had experienced a head injury 8 years previously, with recurrent severe epistaxis. An ICA aneurysm was detected on computed tomography. The trapping and bypass was planned. However, sudden epistaxis occurred, we performed trapping to stop the bleeding and save his life. After the operation, no right ICA or aneurysm was detected. However, severe epistaxis recurred two months after the operation. In the second operation, a ligation of the common -/- external carotid artery and a severance of an ICA portion between the ophthalmic artery and the aneurysm were insufficient to stop the bleeding. This case indicates ICA trapping, even if a trapping portion is below an ophthalmic artery, is insufficient to treat an ICA aneurysm. ICA aneurysms should be suspected when a patient present with recurrent -/- massive epistaxis, who has a head injury history, even if it is far past.

Spontaneous Intraventricular Pneumocephalus.

BACKGROUD: Pneumocephalus without a known underlying cause is defined as spontaneous pneumocephalus. Few patients of intraventricular pneumocephalus have been reported. PATIENT PRESENTATION: An 84-year-old man presented with dysarthria and incontinence. Computed tomography revealed an intraventricular pneumocephalus, thinning in the petrous bone, fluid in the air cells, and cleft in temporal lobe. A right subtemporal extradural approach was taken to detect bone-/-dural defects, and a reconstruction was performed using a musculo-pericranial flap. CONCLUSION: This is the first patient of an isolated intraventricular spontaneous pneumocephalus without any other site air involved. Surgical approaches to repair such bone and dura defects should be considered an appropriate option.

Histamine H2-Blocker and Proton Pump Inhibitor Use and the Risk of Pneumonia in Acute Stroke: A Retrospective Analysis on Susceptible Patients.

BACKGROUND: Although histamine H2-blockers (H2B) and proton pump inhibitors (PPI) are used commonly to prevent gastrointestinal bleeding in acute stroke, they are implicated in the increased risk of pneumonia in other disease populations. In acute stroke, the presence of distinctive risk factors of pneumonia, including dysphagia and impaired consciousness, makes inclusive analysis vulnerable to confounding. Our aim was to assess whether acid-suppressive drugs increase pneumonia in acute stroke in a population controlled for confounding. METHODS: We analyzed acute stroke patients admitted to a tertiary care hospital. To minimize confounding, we only included subjects who could not feed orally during 14 days of hospitalization. Exposure was defined as H2B or PPI, given in days; the outcome was development of pneumonia within this period. The incidence was calculated from the total number of pneumonias divided by the sum of person-days at risk. We additionally performed multivariate Poisson regression and propensity score analyses, although the restriction largely eliminated the need for multivariate adjustment. RESULTS: A total of 132 pneumonias occurred in 3582 person-days. The incidence was 3.69%/person-day (95% confidence interval (CI); 3.03-4.37%/day). All subjects had dysphagia. Stroke severity and consciousness disturbances were well-balanced between the groups exposed to H2B, PPI, or none. The relative risk (RR) compared with the unexposed was 1.22 in H2B (95%CI; 0.83-1.81) and 2.07 in PPI (95% CI; 1.13-3.62). The RR of PPI compared with H2B was 1.69 (95%CI; 0.95-2.89). In multivariate regression analysis, the RRs of H2B and PPI were 1.24 (95% CI; 0.85-1.81) and 2.00 (95% CI; 1.12-3.57), respectively; in propensity score analyses they were 1.17 (95% CI; 0.89-1.54) and 2.13 (95% CI; 1.60-2.84). CONCLUSIONS: The results of this study suggested that prophylactic acid-suppressive therapy with PPI may have to be avoided in acute stroke patients susceptible to pneumonia.

Delayed Acute Subdural Hematoma Associated With Percutaneous Coronary Intervention.

BACKGROUND: Delayed acute subdural hematoma (DASH) is a subdural hematoma which is detected later. An initial computed tomography (CT) does not reveal any intracranial hemorrhage at all. Few patients of DASH after mild traumatic brain injury associated with percutaneous coronary intervention (PCI) have been published. PATIENT PRESENTATION: A 63-year-old woman presented with cardiac pulmonary arrest due to acute myocardial infarction and lethal arrhythmia. She had hit her head on the road. The initial CT did not reveal any hemorrhage in the intra-cranium. She fully recovered after PCI. However, 1 hour after PCI, she lost consciousness and immediate CT showed acute subdural hematoma and subarachnoid hemorrhage. The period from losing consciousness to brain herniation presenting as anisocoria was very short-only 30 minutes in our patient. Although emergent evacuation of hematoma and external decompression were performed, the patient died 1 day after the operation. CONCLUSION: The authors encountered a patient of DASH after PCI that resulted in death. Clinicians should be aware that subdural hemorrhage can occur after PCI if no hemorrhage is noted in the initial head CT, and the operation should be performed as soon as possible when the consciousness level decreases.

Malignant isolated cortical vein thrombosis with type II protein S deficiency: a case report.

BACKGROUND: The incidence of cerebral venous thrombosis (CVT) is low, and in particular, isolated cortical vein thrombosis (ICVT) is very rare. The diagnosis of ICVT is difficult by using conventional computed tomography (CT) and magnetic resonance imaging (MRI). However, with appropriate treatment, ICVT has a good prognosis. CASE PRESENTATION: Herein, we present a rare case of a 40-year-old woman with ICVT and type II protein S (PS) deficiency, who experienced a stroke. She initially presented with generalized convulsions. A CT scan showed intracerebral hemorrhage (ICH) in the left temporoparietal region. However, her condition rapidly deteriorated and she went into a coma approximately 20 h after admission. A second CT scan revealed significant ICH expansion and transfalcine herniation. Decompressive hemicraniectomy with duraplasty was performed, and ICVT was confirmed owing to abnormal vascular tone and black appearance of the cortical vein. She underwent anticoagulation therapy and rehabilitation, and gradually recovered. CONCLUSION: We experienced an extremely rare case of isolated cortical vein thrombosis related with type II PS deficiency. CT-digital subtraction angiography is a useful supportive technique in the diagnosis of ICVT. Decompressive hemicraniectomy is effective for hemorrhage extension cases, and ICVT with hemorrhage might require early anticoagulation therapy.

Progressive Intracranial Vertebral Artery Dissection Presenting with Isolated Trigeminal Neuralgia-Like Facial Pain.

Intracranial vertebral artery dissection (IVAD) is a potentially life-threatening disease, which usually presents with ischemic stroke or subarachnoid hemorrhage. IVAD presenting with isolated facial pain is rare, and no case with isolated trigeminal neuralgia- (TN-) like facial pain has been reported. Here, we report the case of a 57-year-old male with IVAD who presented with acute isolated TN-like facial pain that extended from his left cheek to his left forehead and auricle. He felt a brief stabbing pain when his face was touched in the territory of the first and second divisions of the left trigeminal nerve. There were no other neurological signs. Magnetic resonance imaging (MRI) of the brain 7 days after onset revealed dissection of the left intracranial vertebral artery without brain infarction. The pain gradually disappeared in approximately 6 weeks, and the patient remained asymptomatic thereafter, except for a brief episode of vertigo. Follow-up MRI revealed progressive narrowing of the artery without brain infarction. This case indicates that IVAD can present with isolated facial pain that mimics TN. IVAD should be considered in the differential diagnosis of acute facial pain or TN.

[A case of glomerulonephritis associated with ventriculo-atrial shunt].

Nephritis associated with a chronically infected ventriculo-atrial (VA) or ventriculo-peritoneal (VP) shunt is known as shunt nephritis. A 60-year-old woman who had a VA shunt implanted for hydrocephalus began to show intermittent low-grade fever of an unknown origin, as well as hypertension and lower leg edema. Laboratory findings showed renal insufficiency with proteinuria, and a percutaneous renal biopsy was performed. Light microscopy revealed findings of membranoproliferative glomerulonephritis. Shunt nephritis was suspected. The VA shunt was removed and the VP shunt was replaced subsequently. Signs of renal impairment were recovered after surgery. However, a new VA shunt was implanted because of a shunt malfunction. Shunt nephritis is a rare complication associated with shunt system implantation. It can be treated successfully by removing the shunt system immediately. It should be considered that shunts, especially VA shunts, always carry a risk of nephritis.

[A case of multifocal fibrosclerosis presenting with chronic subdural hematoma].

Multifocal fibrosclerosis(MFS) is a rare disorder of unknown etiology, characterized by chronic inflammation with dense fibrosis and lymphoplasmacytic infiltration into the connective tissue of various organs. Recently, MFS was classified as IgG4-related systemic disease. In this paper, we report a 60-year-old man with no history of head injury presenting with chronic subdural hematoma(CSDH). After surgery, he complained of severe, continuous headache and persistent high-grade fever. Extensive evaluation, including 67Ga scintigraphy suggesting inflammations in various organs, liver needle biopsy showing sclerosing cholangitis, and blood examination showing elevated serum IgG4 levels, led to the diagnosis of MFS. To our knowledge this is the first report of MFS causing CSDH. The mechanism of the formation of CSDH is presumed to involve reactive granular membrane together with exudative subdural collection caused by MFS, which gives rise to minor and repeated bleeding. In this case, oral corticosteroid therapy was dramatically effective in the treatment of the condition.

[Association between phenytoin-induced gingival hyperplasia and periodontopathic bacteria in institutionalized patients with severe motor and intellectual disabilities].

The association between phenytoin-induced gingival hyperplasia (PIGH) and five major periodontopathic bacteria was investigated by real-time PCR in 12 subjects (30.6 +/- 7.95 years of age) with severe motor and intellectual disabilities. The subjects had not received antibiotics or periodontal therapy within three months. A healthy gingival site or a site with the lowest gingival hyperplasia score (minimum site), and a site with the maximum score of gingival hyperplasia (maximum site) on each subject were selected for investigation. The clinical parameters were measured after microbial sampling. The following results were obtained: 1. The detection rates of both Treponema denticola and Porphyromonas gingivalis were 75% (9/12) at the maximum sites, and that of Tannerella forsythia was 33.3% (4/12). The detection rate of Prevotella intermedia was 58.3% (7/12), but no significant correlation was observed between this bacterium and the other types of bacteria in quantitative analysis. 2. The detection rate of P. gingivalis with type II fimA was 25% (1/4) at the minimum sites, and 83.3% (5/6) at the maximum sites. 3. The samples were divided into three groups; namely, those with no detection of P. gingivalis at either the minimum sites or the maximum sites, no detection at the minimum sites but detection at the maximum sites, and detection at both sites. In conclusion, 1) the detection rates of T. denticola and P. gingivalis were the highest at the maximum sites. In particular, the detection rate of P. gingivalis with type II fimA was high in the present study, and 2) the subgingival microflora was diverse because of the diverse severity of gingival inflammation associated with PIGH.

Single photon emission computed tomography of reversible magnetic resonance imaging abnormalities of deep cerebral venous thrombosis accompanied with malignant glioma.

BACKGROUND: Deep cerebral venous thrombosis with reversible MRI findings in bilateral thalamus have been scarcely described in the literature, but none mentioned the SPECT findings such as in our case. CASE DESCRIPTION: A 43-year-old woman presented with cerebral venous thrombosis accompanied with malignant glioma. T(2)- and diffusion-weighted MRI showed high intensity with a mass lesion in the left frontal lobe and mild high and high intensity in the right frontal lobe, parietal lobe, and bilateral thalamus. Cerebral angiography showed occlusions of the SSS and the DCVs. Furthermore, the initial SPECT showed apparent hypoperfusion in the same lesions on MRI. After the treatment involving infusion therapy and surgery, she recovered with a mild cognitive deficit. Although the repeat T(2)- and diffusion-weighted MRI revealed the disappearance of high intensity, the repeat SPECT showed the persistent hypoperfusion in the infarcted lesions. CONCLUSION: There might be a discrepancy between MRI and SPECT findings of the reversible case of DCVT, and SPECT also could be useful for the evaluation of the amelioration of DCVT.

[Immunohistochemical study is helpful for the diagnosis of cerebellar clear cell ependymoma with atypical radiological findings--case report].

A 67-year-old male presented with a clear cell ependymoma with symptoms of ataxic gait and dizziness. Magnetic resonance imaging showed a ring enhanced and circumscribed mass lesion with some cysts in the left cerebellar hemisphere, and the vertebral artery angiogram showed the vascurality of the tumor fed by both the left posterior inferior cerebellar artery and the left superior cerebellar artery mainly. They demonstrated suspicious finding of metastatic tumor, glioblastoma, or cystic meningioma. Surgery via the left suboccipital approach revealed a whitish and solid tumor, which was demarcated from the cerebellar parenchyma and had no continuity with the 4th ventricle. Total resection of the tumor was successfully performed. The hematoxilyn-eosin staining of the surgical specimen was similar to hemangioblastoma or oligodendroglioma, however, immunohistochemical findings for glial fibrillary acidic protein, vimentin, epithelial membrane antigen, and factor VIII were compatible with clear cell ependymoma. The patient's postoperative course was uneventful, and his symptoms improved. Clear cell ependymoma is known as a variant of ependymoma, which is usually located at the foramen of Monro. We think that the immunohistochemical study is highly helpful for the diagnosis of the cerebellar tumor with atypical presentation such as our case.

[Speech disturbance following myelography with iohexol: a case report].

Iohexol (Omnipaque) is a non-ionic contrast media for neuroradiology, which causes the neurological complications on rare occasions. A 63-year-old, righthanded female suffered from dysphemia 12 hours after myelography with iohexol via lumbar puncture. The initial cerebral computed tomography scan revealed the generalized contrast media uptake into the cisterns, which was typical to the finding after intrathecal iohexol injection. Although the cerebral magnetic resonance (MR) imaging 7 days after attack also demonstrated no abnormal finding, the electrical encephalogram revealed the sporadic sharp wave activity. Her nonfluent speech deficit had recovered gradually, however, the neurogenic stuttering was still remained. Some literatures previously reported the cases with speech deficits as complications of metrizamide myelography, whereas, a case manifesting as speech disturbance following myelography with iohexol has been not reported. Metrizamide is also a non-ionic agent and had a lower incidence of speech disturbance, which is supposed to be associated with a focal superficial neurotoxic reaction of the cerebrum. Therefore, our unfortunate case suggests that iohexol could rarely cause speech disturbance such as metrizamide.

[Calcified chronic subdural hematoma complicated with subcortical hemorrhage: case report].

The patient was a 46-year-old male, who suffered from mild head trauma in January 2002, and general convulsions with unconsciousness on February 28. Slight right hemiparesis and aphasia were presented after the epilepsy attack. CT scan revealed a large lesion of mixed density occupying the left temporal space. It showed linear high density in its medial margin and had compressed the left temporal lobe strongly, causing mid-line shift. The lesion was suspected to be a calcified chronic subdural hematoma and the patient was admitted to our hospital on February 28. The symptoms had improved the next day but they began to get worse again gradually after admission. T1-weighted MR image showed high intensity areas under the subdural hematoma, which were suspected to be subcortical hemorrhage. Six days after admission, consciousness disturbance became progressive. The calcified hematoma had not enlarged but brain edema had increased. On CT, an operation was performed and the calcified old hematoma and the new subdural hematoma surrounding it were removed. The diagnosis of organized chronic subdural hematoma was made at the time of the operation. The contents of this calcified subdural hematoma was mostly old dark-gray substance, but some fresh bleeding point was seen at the inner surface of the outer membrane. At the bottom there was a hard, calcified layer which adhered tightly to the brain. Adhesion between the inner membrane of the hematoma and brain surface which related to the subcortical hemorrhage was presented. It seemed impossible to remove the inner membrane without damaging the brain so no attempt was made to do so. The aphasia and right hemiparesis improved 3 weeks after the operation and the patient was discharged on April 4. He has no neurological deficits and is under periodic observation. A calcified chronic subdural hematoma has rarely been encountered and the etiology, imaging diagnosis, and management are unclear. We presented the interesting image findings on this case and discussed the etiology of this disease.

[Aseptic meningitis as a complication caused by an allergic reaction after microvascular decompression: two case reports].

We report here two cases of patients complicated with aseptic meningitis after microvascular decompression (MVD). The first case, a 56-year-old female complained of headache with high fever 18 days after the MVD for right trigeminal neuralgia. The amount of cells in cerebrospinal fluid (CSF) had so much increased that bacterial meningitis was suspected. However, there was no improvement after antibiotics therapy, so immune globulin was injected and the meningitis gradually improved. Eosinophilia remained in peripheral blood and the symptoms improved rapidly after the steroid therapy. Because of this, we suspected that meningitis was caused by an abnormal allergic reaction. The second case, a 30-year-old male complained of headache with mild fever 15 days after MVD for left hemifacial spasm. The amount of cells in CSF increased, so bacterial meningitis was suspected. Eosinophilia remained in peripheral blood and the steroid therapy proved very effective for the meningitis. Because of this, we suspected that meningitis was caused by an abnormal allergic reaction. We suspected that the two patients suffered from aseptic meningitis caused by allergic reaction, and the antigen for this abnormal allergic reaction was the foreign materials used for MVD. The materials were Dacron for prostesis, Goatex or Lyodula for dural plasty, fibrin glue for preventing CSF leakage. We ascertained that the abnormal allergic reaction was caused by human fibrinogen in the second case. It is important to be aware of such allergic reaction to fibrin glue in the post-operative stage after MVD.

[A dissecting aneurysm of the posterior inferior cerebellar artery was reduced spontaneously during conservative therapy: case report].

We report here a case of a patient with a dissecting aneurysm of the anterior medullary segment of the posterior inferior cerebellar artery (PICA) which presented with Wallenberg's syndrome. A 32-year-male presented with an unusual case of Wallenberg's syndrome due to a dissecting aneurysm of the PICA manifesting as a sensation of heaviness in the occipital region and vertigo. The occipital symptoms persisted and vertigo and vomiting developed after 6 days. Numbness developed on the left side of the patient's face, and hyperalgesia on the right side of the body. The diagnosis of Wallenberg's syndrome was based on the above findings. MRI revealed infarction of the lateral aspect of the medulla oblongata and MR angiography revealed dilatation in the proximal portion of the left PICA. Digital subtraction angiography revealed that the left vertebral artery was essentially normal, but there was a spindle-shaped dilatation in the proximal portion of the left PICA. We carried out conservative therapy at the patient's request and 3D-CTA revealed that the dissecting aneurysm was markedly reduced in size seven months after the onset. Dissecting aneurysms of the intracranial posterior circulation have been shown to be less uncommon than previously thought. However, those involving the PICA without involvement of the vertebral artery at all are extremely rare. The natural history of the dissecting PICA aneurysm was unknown, and the indication for surgical treatment of such aneurysms remains controversial. Management options are conservative treatment, open surgical treatment including wrapping, trapping, and resection with reconstruction, but almost all of the patients underwent radical treatment to prevent rupture of the aneurysm. However we had no knowledge of the risk of rupture of a PICA dissecting aneurysm presenting with ischemic symptoms. We have reviewed the well-documented 15 cases of dissecting aneurysms of the PICA reported in the literature and we discuss the management of the dissecting PICA aneurysm presenting with ischemic symptoms.