Neurologia medico-chirurgica
Online ISSN : 1349-8029
Print ISSN : 0470-8105
ISSN-L : 0470-8105
Volume 53, Issue 10
Displaying 1-16 of 16 articles from this issue
Original Articles
  • Satsuki MIYATA, Masashi URABE, Akira GOMI, Mutsumi NAGAI, Takashi YAMA ...
    2013 Volume 53 Issue 10 Pages 645-654
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Cytosolic isocitrate dehydrogenase 1 (IDH1) with an R132H mutation in brain tumors loses its enzymatic activity for catalyzing isocitrate to α-ketoglutarate (α-KG) and acquires new activity whereby it converts α-KG to 2-hydroxyglutarate. The IDH1 mutation induces down-regulation of tricarboxylic acid cycle intermediates and up-regulation of lipid metabolism. Sterol regulatory element-binding proteins (SREBPs) regulate not only the synthesis of cholesterol and fatty acids but also acyclin-dependent kinase inhibitor p21 that halts the cell cycle at G1. Here we show that SREBPs were up-regulated in U87 human glioblastoma cells transfected with an IDH1R132H-expression plasmid. Small interfering ribonucleic acid (siRNA) for SREBP1 specifically decreased p21 messenger RNA (mRNA) levels independent of the p53 pathway. In IDH1R132H-expressing U87 cells, phosphorylation of Retinoblastoma (Rb) protein also decreased. We propose that metabolic changes induced by the IDH1 mutation enhance p21 expression via SREBP1 and inhibit phosphorylation of Rb, which slows progression of the cell cycle and may be associated with non-aggressive features of gliomas with an IDH1 mutation.
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  • Makoto IDEGUCHI, Koji KAJIWARA, Koichi YOSHIKAWA, Hirokazu SADAHIRO, S ...
    2013 Volume 53 Issue 10 Pages 655-662
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Abnormal hemodynamics during extirpation of a para-medulla oblongata (MO) tumor is common and may be associated with direct vagal stimulation of the medullary circuit. However, resection of tumors on the dorsal MO may also induce hemodynamic instability without direct vagal stimulus. The objective of this study was to examine the characteristics of hemodynamic instability unrelated to vagal stimulus during dissection of an intra-fourth ventricular tumor with attachment to the dorsal MO. A retrospective analysis was performed in 13 patients. Abnormal hemodynamics were defined as a > 20% change from the means of the intraoperative mean arterial pressure (MAP) and heart rate (HR). Relationships of intraoperative hemodynamics were evaluated with various parameters, including the volume of the MO. Six patients (46.2%) had intraoperative hypertension during separation of the tumor bulk from the dorsal MO. The maximum MAP and HR in these patients were significantly greater than those in patients with normal hemodynamics (116.0 ± 18.0 mmHg versus 85.6 ± 6.5 mmHg; 124.3 ± 22.8 bpm versus 90.5 ± 14.7 bpm). All six cases with abnormal hemodynamics showed hemodynamic fluctuation during separation of the tumor bulk from the dorsal MO. The preoperative volume of the MO in these patients was 1.11 cc less than that in patients with normal hemodynamics, but the volume after tumor resection was similar in the two groups (5.23 cc and 5.12 cc). This suggests that the MO was compressed by the conglutinate tumor bulk, with resultant fluctuation of hemodynamics. Recognition of and preparation for this phenomenon are important for surgery on a tumor located on the dorsal MO.
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  • Da LI, Shu-Yu HAO, Zhen WU, Gui-Jun JIA, Li-Wei ZHANG, Jun-Ting ZHANG
    2013 Volume 53 Issue 10 Pages 663-675
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    To evaluate the long-term outcome and functional recovery of intramedullary medullocervical ependymoma (IME), the clinical charts of 38 surgically treated consecutive cases of IME were reviewed. Follow-up was obtained prospectively. The mean age of the patients (19 male and 19 female) was 35.3 years (range: 11–60 years). Complete resection was achieved in 33 (86.8%) patients. Fourteen patients worsened postoperatively; five and seven of these improved to their baseline levels within 1 and 3 months, respectively. By 1 year postoperatively, 17 patients returned to work. After a mean follow-up duration of 81.5 months, 31 patients improved or stabilized, and 3 had recurrence. The means of the modified McCormick grade (mMG) scores before the operation, at discharge, 1 year after the operation, and at the most recent evaluation were 1.76, 2.13, 1.82, and 1.84, respectively. A favorable long-term outcome of the mMG was associated with a good preoperative status (mMG I) (odds ratio [OR] = 9.956, p = 0.008) and well-defined tumor boundary (OR = 7.829, p = 0.035). Improvements in the postoperative walking dysfunction and paresthesia over time were associated with the absence of preoperative walking dysfunction (p = 0.047) and paresthesia (p = 0.028), respectively. The 12-year progression/recurrence-free survival and overall survival rates were 92.0% and 93.7%, respectively. The study suggests that the goal of surgery is to stabilize the preoperative neurological function and that a favorable outcome may be achieved in patients with good preoperative statuses and well-defined tumor boundaries. Surgery should be performed as soon as possible after the diagnoses and before the neurological functions deteriorate.
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  • Kota KAGAWA, Koji IIDA, Akiyoshi KAKITA, Masaya KATAGIRI, Takeshi NISH ...
    2013 Volume 53 Issue 10 Pages 676-687
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Based on intracranial-video electroencephalography (EEG), histopathological features, and postoperative seizure outcome, we elucidated the epileptogenicity in patients with dysembryoplastic neuroepithelial tumor (DNT). Five patients (P1–P5) pathologically diagnosed with DNT underwent intracranial-video EEG to identify the ictal onset zone and irritative zone. We evaluated the correlations of ictal onset zone and irritative zone with the magnetic resonance imaging-visible lesion (MRI-lesion) and their histopathological features. Intracranial-video EEG located the ictal onset zone adjacent to the MRI-lesion margin in four patients with complex/simple forms of DNT subcategory, and on the MRI-lesion in P3 with a nonspecific DNT form. The irritative zone extended to surrounding regions of the ictal onset zone in all patients. Histopathologically, MRI-lesions were characterized by specific glioneuronal elements, whereas the ictal onset zone and irritative zone were represented with dysplastic cortex accompanying oligodendroglia-like cells in four (P1, P2, P4, and P5) of five patients. Cortical dysplasia was identified with typical histopathologic features in the irritative zone remote from the MRI-lesion in P5. P3, with a nonspecific form, indicated prominent component of dysplastic cortex with oligodendroglia-like cells scattered in the MRI-lesion. Lesionectomy of MRI-lesion with additional cortical resections (including the ictal onset zone and irritative zone) yielded postoperative seizure freedom (Engel Class I) in P3, P4, and P5, while P1 and P2 (with only lesionectomy) experienced postoperative residual seizure (Class II and III in each patient). Our results suggest the intrinsic epileptogenicity of DNT. The topographical correlation indicated that the dysplastic cortex accompanying oligodendroglia-like cells was more epileptogenic than the specific glioneuronal elements itself. Meticulous intracranial-video EEG analysis delineating the MRI nonvisible ictal onset zone and the irritative zone may yield better seizure outcome.
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  • Akira NAKAMIZO, Megumu MORI, Daisuke INOUE, Toshiyuki AMANO, Masahiro ...
    2013 Volume 53 Issue 10 Pages 688-694
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Although many investigators have reported the hearing function in the immediate postoperative period in patients with vestibular schwannoma (VS), little is known about the long-term outcomes of the postoperative hearing. The aim of this study was to analyze the long-term hearing outcomes at a mean follow-up of 5 years in patients with unilateral VS treated via the retrosigmoid approach. Twenty-four patients with immediate postoperative serviceable hearing who underwent repeated audiogram or phone interview were included in this study. During the mean follow-up period (68.8 ± 30.2 months, range 14–123 months), serviceable hearing was preserved in 20 out of the 24 patients (83%). Pure tone average (PTA) was reevaluated within 6 months in seven patients. In the two patients whose PTA deteriorated ≥ 5 dB in 6 months after surgery, their PTA worsened ≥ 15 dB compared to the immediate postoperative PTA. In the remaining five patients whose PTA deteriorated < 5 dB in 6 months after surgery, PTA was maintained within a 15-dB deterioration at the final follow-up (p = 0.04, Fisher’s exact test). According to Kaplan–Meier survival plots, the 5-year or 7-year preservation rate of serviceable hearing was 86.2% or 71.8%, respectively. Further study will be needed to clarify the mechanism underlying the long-term decline of serviceable hearing; however, the deterioration of PTA in the early postoperative period may help to predict the long-term outcomes of hearing.
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Case Reports
  • Katsuya MASUI, Taiji YONEZAWA, Yukei SHINJI, Ryo NAKANO, Seisuke MIYAM ...
    2013 Volume 53 Issue 10 Pages 695-698
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Melanoma metastases to the pituitary gland are extremely rare, with only a few reported cases. We report an unusual case of pituitary metastatic melanoma in which the patient presented with pituitary apoplexy. A 68-year-old man presented general fatigue and anorexia following sudden headache. Neurological examination disclosed bitemporalhemianopsia. Computed tomography (CT) scans revealed a suprasellar mass including intratumoral hematoma. Magnetic resonance (MR) images demonstrated a circumscribed mass lesion in the intra- and suprasellar regions, compressing the optic chiasm. Surgical exploration was performed through a transsphenoidal approach, and a mixture of tumor and necrotic tissue with old hematoma was obtained. The histological examination of the specimen revealed a partly necrotic, malignant tumor with focal melanotic pigmentation. Histopathologically, the diagnosis was consistent with pituitary apoplexy caused by hemorrhage from pituitary metastatic melanoma.
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  • Shinya ICHIMURA, Koichi HARA, Reiko SHIMOKAWA, Hiroshi KAGAMI, Makoto ...
    2013 Volume 53 Issue 10 Pages 699-702
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 24, 2013
    JOURNAL OPEN ACCESS
    Both intraosseous and microcystic meningiomas are rare tumor types. We report the case of a 66-year-old woman with intraosseous microcystic meningioma without a mass lesion. She presented with a rare intraosseous microcystic meningioma manifesting as pain. Radiological examination revealed an osteolytic lesion in the right parietal bone. Magnetic resonance (MR) images showed iso- to hypointensity on T1-weighted images and hyperintensity on T2-weighted images corresponding to the lesion. T1-weighted MR imaging with gadolinium enhancement better defined the marginal area. The inner table of the skull was disrupted prominently, and both sides of the outer table were eroded. There was fluid leakage during surgery but no obvious tumor mass. Histological examination revealed microcystic meningioma in the inner part of the defective bone. A macroscopic lesion was not found, because most of the tumor comprised microcysts, and their contents leaked out during the surgical procedure. Intraosseous microcystic meningioma may be considered as one of the differential diagnoses when the intraosseous tumor in the skull has fluid leakage and does not have a mass lesion during the surgery.
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  • Naoki NITTA, Tadateru FUKAMI, Kazuhiko NOZAKI
    2013 Volume 53 Issue 10 Pages 703-706
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    We present a third reported case of intracranial germinomas occurring in two brothers. They underwent chemotherapy and radiotherapy after biopsy and are doing well for 10 years and 20 months, respectively. The pertinent literatures are reviewed, and the possible role of heredity in the pathogenesis of familial occurrence of germinomas is discussed.
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  • Toshihide TANAKA, Naoki KATO, Ken AOKI, Mitsuyoshi WATANABE, Takao ARA ...
    2013 Volume 53 Issue 10 Pages 707-711
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    This report describes a rare case of a patient with growth hormone (GH)-secreting pituitary adenoma with malignant transformation resulting in multiple metastases to the dura mater of the cerebral convexity and high cervical spine. The patient was a 60-year-old man with a previous history of pituitary adenoma with suprasellar extension who had undergone transsphenoidal surgery, craniotomy for a convexity tumor, and suboccipital craniotomy for a cerebellar tumor. Thirteen years after the initial surgery, suboccipital craniotomy for a cervicomedullary junction tumor and cervicospinal surgery for a metastatic tumor was performed. Histologic findings of resected specimens demonstrated that the primary pituitary tumor was typical adenoma (similar to specimens from the initial surgery) but that the cerebellar and the dural tumor from the high cervical spine had a high incidence of mitotic figures, and cellular anaplasia with nuclear polymorphism and necrosis. In addition, the serum levels of GH were noted to have decreased with recurrence of the tumor. It was concluded that patients with pituitary adenoma, even when benign, must be carefully followed for signs of malignant transformation, and spinal or distant metastases.
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  • Mário Henrique Girão FARIA, Ricardo Henrique DÓRIA-NETTO, Gustavo Jun ...
    2013 Volume 53 Issue 10 Pages 712-716
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Melanotic schwannoma (MS) is an unusual variant of nerve sheath neoplasm. Only 10% of these tumors will undergo malignant degeneration, with exceedingly rare reported metastasis. We present a 32-year-old woman with a 6-month history of cervical pain and left arm progressive weakness. Neurological examination showed a left upper limb radicular pain, with pyramidal syndrome at C5 level. The magnetic resonance imaging (MRI) study highlighted an intradural extramedullary heterogeneous mass along the spinal cord at the C4–C5 level, slightly hyperintense with T1 and hypointense with T2-weighted sequences, invading the left neural foramen. The patient underwent C3–C5 laminectomy with total resection of a black tumor. In the postoperative period, a patent deficit of shoulder abduction ensued related to the nervous section. Microscopically, compactly fascicles of spindle-shaped cells with pleomorphic and hypercromatic nuclei, dark brown intracellular pigments, as well as some mitotic figures were seen. Immunohistochemical stains for S-100, Human Melanoma Black-45 (HMB-45), and vimentin were positive, with Ki-67 Labelling Index (LI) of 15% compatible with MS. Six months after radiotherapy she presents local recurrence and lung metastatic dissemination of the MS. She underwent left pulmonary segmentectomy, followed by chemotherapy and radiosurgery. The patient developed a febrile neutropenia and worsening of general status, and died after 3 months due to respiratory complications. MS are rare tumors with potential for local recurrence and distal metastasis. Complete surgical resection remains as the treatment of choice, once the uncommon cases with malignant progression shows low response to chemo and radiotherapy.
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  • Kenji FUJIMOTO, Jun-ichiro KURODA, Keishi MAKINO, Yu HASEGAWA, Jun-ich ...
    2013 Volume 53 Issue 10 Pages 717-721
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Skull metastases occur in patients with various malignancies; however, those resulting from intrahepatic cholangiocarcinoma (ICC) have been rarely reported. In our hospital, 324 patients were diagnosed with metastatic brain or skull tumors from June 1969 to June 2011, but only 3 of them (0.9%) developed skull metastases from ICC. We report the case of 3 patients with skull metastases from ICC. A combination of computed tomography (CT), contrast-enhanced magnetic resonance imaging (MRI), 18F-fluorodeoxyglucose positron emission tomography (FDG-PET), and methionine-PET were used for imaging. Sites of tumors were the lateral left orbit and right parietal bone in case 1, the left parietal bone, left temporal bone, and lateral left orbit in case 2, the right petrous bone, right occipital bone, and upper cervical vertebra in case 3. The metastases were confirmed to have originated from ICC by biopsy in two of the cases and diagnosed by MRI and FDG-PET in case 2. Radiosurgery and radiotherapy had positive effects on symptom improvement and cosmetic problems.
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  • Daichi KAWAMURA, Toshihide TANAKA, Michiyasu FUGA, Takeshi YANAGISAWA, ...
    2013 Volume 53 Issue 10 Pages 722-726
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    The report describes a rare case of a patient with a calcified cerebellar metastasis arising from a primary ovarian cancer. The patient was a 33-year-old woman with a long history of stage IIIc ovarian cancer who had undergone transabdominal hysterectomy and bilateral oophorectomy followed by chemotherapy with gemcitabine hydrochloride. Incidentally, computed tomography (CT) revealed a cerebellar tumor with calcification. The size of the tumor gradually increased, and lateral suboccipital craniotomy was performed for gross total removal of the tumor. The histological diagnosis was ovarian mucinous adenocarcinoma. The patient’s postoperative course was uneventful, and she was discharged two days after surgery. Brain metastases from ovarian cancer are rare. In the review of metastatic brain tumors arising from a primary ovarian cancer in the Department of Obstetrics and Gynecology at our institution, this phenomenon was noted in only 10 cases (0.24%) of 4,158 patients with ovarian cancer seen at our center over a period of 8 years. Moreover, only three cases of calcified metastatic brain tumor have been reported previously. In conclusion, complete tumor resection may be an acceptable approach for patients with calcified metastatic tumors both for therapeutic considerations and to obtain tissue for confirmation of histopathological diagnosis. Metastatic brain tumors can be calcified, and should be considered within the differential diagnosis of calcified intracranial lesions to avoid any delay in diagnosis or treatment.
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  • Satoshi TSUTSUMI, Hiroshi IZUMI, Yukimasa YASUMOTO, Masanori ITO
    2013 Volume 53 Issue 10 Pages 727-729
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    A 67-year-old woman sensed a slowly growing, painless hard mass in the left parietal region. Cranial computed tomography showed focal bony erosion and homogeneous sclerotic change at the affected site. Magnetic resonance (MR) imaging revealed an enhanced subcutaneous mass and irregularly thickened dura mater. Intraoperatively, the subcutaneous tumor was found to be strongly adhered to the temporalis muscle. The outer table was eroded adjacent to the subcutaneous tumor, whereas the bony structures of the inner table were intact. The dura mater underneath had irregular-shaped, yellowish convolutions both on the outer and inner surfaces. The patient underwent total tumor resection with sufficient normal margins. The histological diagnosis was World Health Organization (WHO) grade I meningioma, with finger-like outward extensions through the dura mater and overlying skull, and infiltration among into the temporalis muscle fibers. Meningiomas may form a subcutaneous mass without intracranial growth.
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  • Ryu KUROKAWA, Phyo KIM, Toshiki KAWAMOTO, Hadzki MATSUDA, Shujiro HAYA ...
    2013 Volume 53 Issue 10 Pages 730-734
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Neurocutaneous melanosis (NCM) is a rare condition characterized by central nervous system melanocytic tumors associated with congenital melanocytic nevi. Phacomatosis pigmentovascularis (PPV) is an association of vascular nevus with pigmentary nevus. Aberrant maturation of neural crest-derived cells is considered to be related to pathogenesis in both conditions. However, association of NCM and PPV has not been reported to the best of our knowledge. Melanocytoma, which usually involves the leptomeninges or spinal cord, is extremely rare in the retroperitoneum. We present here a case of a patient with NCM, PPV, and melanocytic tumors in the spinal cord and retroperitoneum, which were treated surgically. A 40-year-old woman had a 2-year history of dysesthesia and weakness in the left leg. History included congenital giant blue nevus-like lesion in the trunk, a port-wine stain in the sacral area, and Caesarean section performed 8 years before, when diffuse pigmentation in the peritoneum was noted. Magnetic resonance (MR) imaging of the spine revealed an intramedullary tumor at T10 level with paramagnetic signal characteristics. The spinal cord tumor was totally removed, and the histological diagnosis was melanocytoma. Three months later, a left retroperitoneal mass with histological features of melanocytic tumor was removed. Neither tumors recurred and the patient stays ambulatory 4 years after the surgery. Multiple subtypes of melanocytic tumors with distinctive features of NCM and PPV can develop simultaneously, mimicking malignant melanoma. Gross total resection of each tumor, when indicated, is beneficial.
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  • Tao XIE, Xiaobiao ZHANG, Fan HU, Xuejian WANG, Jian WANG, Yong YU, Lin ...
    2013 Volume 53 Issue 10 Pages 735-739
    Published: 2013
    Released on J-STAGE: October 25, 2013
    Advance online publication: September 27, 2013
    JOURNAL OPEN ACCESS
    Hemangioblastoma in the suprasellar region is rare. We present a case of a suprasellar hemangioblastoma that underwent surgical resection using an extended endoscopic transsphenoidal approach. A 64-year-old female patient presented with headache and decreased visual acuity for the last four years, computed tomography (CT) and magnetic resonance imaging (MRI) revealed a 2.5 cm irregular lesion in the suprasellar region. Our preoperative presumptive diagnosis was craniopharyngioma. The patient underwent an extended endoscopic transsphenoidal approach, the mass was subtotally removed. An endoscopic endonasal repair was needed due to the cerebrospinal fluid (CSF) leak. However, 1 month later, the patient got disturbance of consciousness because of the hydrocephalus. Ventriculoperitoneal shunt was used to solve the problem. Pathological findings were compatible with hemangioblastoma. Suprasellar hemangioblastoma is very rare. Any highly vascular lesions located in the suprasellar region should alert the surgeon to the possibility of hemangioblastoma. Extended endoscopic transsphenoidal approach adopted by us should not be the first choice of the treatment procedure for this kind of large and vascular tumor.
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