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Effectiveness of an epidural blood patch for patients with intracranial hypotension syndrome and persistent spinal epidural fluid collection after treatment

Clinical article

Toru Horikoshi, Arata Watanabe, Mikito Uchida, and Hiroyuki Kinouchi

Object

Magnetic resonance imaging may show a fluid collection in the spinal epidural space of patients with spontaneous intracranial hypotension syndrome (SIHS), but the chronological changes remain unclear.

Methods

Brain and spine MR imaging findings were analyzed in 16 patients (9 women and 7 men, mean age 48.6 years) with SIHS before and after treatment.

Results

Diffuse dural enhancement was seen in 15 patients, and the epidural fluid collection in the spinal canal was clear in 15 and equivocal in 1. Symptoms disappeared after bed rest in 1 patient, and an epidural blood patch was performed in 15 patients, resulting in complete resolution of symptoms in 13. After the follow-up period (range 1–20 months, mean 5.0 months), 1 patient had persistent mild headache that gradually worsened in the afternoon, and another patient complained of heaviness of the eyes. Follow-up MR imaging demonstrated disappearance of the dural enhancement in all patients, but a fluid collection in the spinal canal remained in 4. Two of the 4 patients had persistent symptoms, but the other patients exhibited complete resolution of the symptoms.

Conclusions

An epidural blood patch is effective for sealing of CSF leaks, but the resolution of SIHS-related symptoms does not always imply complete eradication of the leakage.

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Anterior paraclinoid aneurysms

Roberto C. Heros

Open access

Spinal glomus AVM presenting solely with groin pain: illustrative case

Natsumi Baba, Ryo Horiuchi, Takashi Yagi, Kazuya Kanemaru, Hideyuki Yoshioka, and Hiroyuki Kinouchi

BACKGROUND

Spinal glomus arteriovenous malformations (AVMs) are rare and can cause neurological morbidity due to spinal hemorrhage, venous hypertension, or mass effect.

OBSERVATIONS

The authors presented a rare case of spinal glomus AVM presenting with groin pain due to nerve root compression by a feeder aneurysm. A 41-year-old woman was referred to the hospital with initial right groin pain that had worsened over 2 months. Magnetic resonance imaging showed intra- and extramedullary abnormal flow voids at the T11–12 level, and spinal angiography revealed an intramedullary AVM, with extramedullary protrusion of an aneurysm on the feeder vessel, which arose from the sulcal artery of the anterior spinal artery. Because compression of the right L1 nerve root by the aneurysm was the likely cause of the patient’s pain, endovascular embolization was performed. The feeder aneurysm disappeared after partial n-butyl 2-cyanoacrylate embolization, and the groin pain disappeared immediately after treatment. Her clinical status has been stable with no recurrence during 1 year of follow-up.

LESSONS

This is the first report of glomus-type AVM presenting with radiculopathy alone. One should not overlook the possibility of spinal AVM among patients with groin pain.

Open access

Hemorrhagic cervical juxta-facet cyst presenting with Brown-Séquard syndrome: illustrative case

Shigeyoshi Shimura, Ryu Saito, Takashi Yagi, Hideyuki Yoshioka, and Hiroyuki Kinouchi

BACKGROUND

Intraspinal juxta-facet cysts of the spine are known to predominate at the lumbar level and is relatively rare at the cervical level. Most cervical spinal lesions are found incidentally, but they sometimes cause myelopathy or radiculopathy in a chronic course.

OBSERVATIONS

The authors present a rare case of hemorrhagic cervical juxta-facet cyst presenting with Brown-Séquard syndrome. An 86-year-old woman presented with acute-onset right hemiparesis following neck pain and was admitted to the local hospital. She was started on antithrombotic therapy with a suspected diagnosis of cerebral infarction, but quadriplegia progressed 2 days later. Cervical magnetic resonance imaging revealed an intraspinal mass at the C4–5 level and she was referred to the authors’ hospital. Her neurological findings on admission revealed right Brown-Séquard syndrome. In emergency surgery, the mass was resected with a posterior approach. Pathological findings showed hemosiderin deposition and fibroblast proliferation, consistent with a juxta-facet cyst with intracystic hemorrhage. The patient recovered well and returned to an independent daily life.

LESSONS

Rarely, juxta-facet cyst of the cervical spine can cause acute Brown-Séquard syndrome due to intraspinal hemorrhage. In a case of hemiparesis that develops following neck pain, hemorrhagic cervical juxta-facet cyst should be taken into consideration as a differentiation.

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Effects of FABP7 on functional recovery after spinal cord injury in adult mice

Nobuo Senbokuya, Hideyuki Yoshioka, Takashi Yagi, Yuji Owada, and Hiroyuki Kinouchi

OBJECTIVE

Elucidating the mechanisms of neuronal injury is crucial for the development of spinal cord injury (SCI) treatments. Brain-type fatty acid–binding protein 7 (FABP7) is expressed in the adult rodent brain, especially in astrocytes, and has been reported to play a role in astrocyte function in various types of brain damage; however, its role after SCI has not been well studied. In this study, the authors evaluated the expression change of FABP7 after SCI using a mouse spinal cord compression model and observed the effect of FABP7 gene knockout on neuronal damage and functional recovery after SCI.

METHODS

Female FABP7 knockout (KO) mice with a C57BL/6 background and their respective wild-type littermates were subjected to SCI with a vascular clip. The expression of FABP7, neuronal injury, and functional recovery after SCI were analyzed in both groups of mice.

RESULTS

Western blot analysis revealed upregulation of FABP7 in the wild-type mice, which reached its peak 14 days after SCI, with a significant difference in comparison to the control mice. Immunohistochemistry also showed upregulation of FABP7 at the same time points, mainly in proliferative astrocytes. The number of surviving ventral neurons in the FABP7-KO mice at 28 days after SCI was significantly lower than that observed in the wild-type mice. In addition, motor functional recovery in the FABP7-KO mice was significantly worse than that of the wild-type mice.

CONCLUSIONS

The findings of this study indicate that FABP7 could have a neuroprotective role that might be associated with modulation of astrocytes after SCI. FABP7 could potentially be a therapeutic target in the treatment of SCI.

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Induction of cyclooxygenase-2 messenger RNA after transient and permanent middle cerebral artery occlusion in rats: comparison with c-fos messenger RNA by using in situ hybridization

Hiroyuki Kinouchi, Haiyen Huang, Shouichi Arai, Kazuo Mizoi, and Takashi Yoshimoto

Object. Recently, two different cyclooxygenase (COX) genes, COX-1 and -2, were identified. In this study, topographic and chronological profiles of COX-2 messenger (m)RNA and c-fos mRNA expression were investigated using in situ hybridization after focal cerebral ischemia.

Methods. Rats undergoing permanent ischemia were decapitated at 30 and 90 minutes and at 2, 4, 8, and 24 hours after middle cerebral artery (MCA) occlusion, and rats undergoing transient ischemia were decapitated at 4, 8, and 24 hours after MCA occlusion that lasted for either 30 or 90 minutes. After brief transient MCA occlusion, c-fos mRNA was induced in the whole MCA territory, adjacent cortex (cingulate cortex), and distant brain regions such as the hippocampus and substantia nigra. In contrast, COX-2 mRNA was not induced in the ischemic core (lateral striatum) but only in the penumbral area (MCA cortex). Long transient and permanent MCA occlusion did not induce c-fos and COX-2 mRNAs in the ischemic core but strongly induced both mRNAs in the penumbral area (medial striatum and periphery of MCA cortex) and adjacent cortex (cingulate cortex). In brain regions distant from the ischemic territory, although c-fos mRNA was induced in the thalamus, substantia nigra, and hippocampus after extended transient and permanent occlusion, COX-2 mRNA was only induced in the bilateral hippocampi. The induction of COX-2 mRNA persisted in all locations even at 24 hours after MCA occlusion.

Conclusions. The distribution of COX-2 mRNA induction was apparently different from that of c-fos mRNA after MCA occlusion. These results pertaining to COX-2 mRNA agree well with the previous observations of changes in prostaglandin metabolism induced by focal cerebral ischemia. However, whether this induction of the COX-2 gene contributes to the histopathological outcome of cerebral ischemia remains to be elucidated.

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Innovative approach in the diagnosis of medulloblastoma in which the 123I-metaiodobenzylguanidine single-positron emission computerized tomography is used

Case illustration

Toshio Sasajima, Hiroyuki Kinouchi, Noriaki Tomura, Jiro Watarai, and Kazuo Mizoi

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Bony carotid canal hypoplasia in patients with moyamoya disease

Clinical article

Arata Watanabe, Tomohiro Omata, Hidehito Koizumi, Shin Nakano, Nobuyasu Takeuchi, and Hiroyuki Kinouchi

Object

The natural history of moyamoya disease is not well known. We have observed that the bony carotid canal is hypoplastic in patients with adult onset moyamoya disease. Bony carotid canal development should represent internal carotid artery (ICA) development, and may stop with the beginning of ICA stenosis. The purpose of this study was to determine the onset of moyamoya disease by measuring the bony carotid canal.

Methods

The normal diameter of the bony carotid canal was evaluated on 4-mm thick bone window CT scans of the skull base in 60 Japanese patients aged 20–80 years, who had minor head trauma or headache considered to be unrelated to the skull base or arterial systems. The relationship between age and bony carotid canal development was assessed in a second group of 50 patients aged 0–19 years, including 10 under 2 years, using CT scans with the same parameters. The diameter of the bony carotid canal in 17 Japanese patients with moyamoya disease was measured.

Results

The normal diameter in adults was 5.27 ± 0.62 mm (mean ± SD). The bony carotid canal developed rapidly before approximately 2 years of age. After fusion of the bony suture, the bony carotid canal developed slowly. The mean diameter of the bony carotid canal was 3.31 ± 0.44 mm in 11 adult patients with adult-onset moyamoya disease. According to the apparent curve of bony carotid canal development, ICA stenosis was assumed to start in early childhood.

Conclusions

Our findings suggest that most cases of Asian moyamoya disease may arise in childhood and that many Asian adult patients with moyamoya disease may develop occlusive vasculopathy in childhood.

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Clear cell meningioma causing Castleman syndrome in a child

Case report

Takashi Sato, Takeshi Sugiyama, Tomoyuki Kawataki, Eiji Sato, Toru Horikoshi, Kanji Sugita, and Hiroyuki Kinouchi

This 11-year-old boy presented with a rare case of Castleman syndrome caused by a clear cell meningioma manifesting as persistent fever of unknown origin, 2 years after glomerulonephritis. Laboratory investigation of the patient showed an increased inflammatory reaction, as well as elevated polyclonal gamma globulin titer and serum level of C-reactive protein. Magnetic resonance imaging revealed a tumor at the cerebellopontine angle. Neurosurgical intervention was performed under the presumptive diagnosis of Castleman syndrome caused by intracranial tumor. Histological examination of the tumor verified that it was clear cell meningioma with infiltration of lymphoplasma cells, and surgical removal resulted in complete resolution of the patient's symptoms and biochemical abnormalities. The present case of clear cell meningioma manifesting as Castleman syndrome shows that the possibility of a brain tumor should be considered in patients presenting with fever of unknown origin, anemia, hypergammaglobulinemia, or other systemic illness.

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A cortical dysembryoplastic neuroepithelial tumor initially occurring in the periventricular white matter

Case report

Tomoyuki Kawataki, Eiji Sato, Tatsuya Kato, Takashi Sato, Toru Horikoshi, and Hiroyuki Kinouchi

In this report, a rare case of dysembryoplastic neuroepithelial tumor (DNET) initially presented as a small white matter lesion with calcification adjacent to the lateral ventricle and extending to the frontal cortex after 7 years. This 1-year-old boy initially suffered from partial seizures. Initial CT revealed a small, low-density area surrounding a tiny calcified mass in the deep white matter of the left frontal lobe. Seven years later, his seizures had become intractable to antiepileptic agents, and MR imaging demonstrated a relatively large mass extending from the calcified lesion up to the adjacent cortical surface. He underwent surgery and the tumor was subtotally removed. Histological examination of the tumor verified it as a DNET consisting of clusters of small oligodendrocytes with floating neurons in the mucoid background. The pattern of the tumor progression in this case suggests that a DNET in the cortex originates from the subependymal germinal layer near the ventricle.