Journal of Clinical Neuroscience (J CLIN NEUROSCI)

Publications in this journal

• Article: Symptomatic brain involvement as the initial manifestation of neuromyelitis optica.

  Ju-Hong Min, Patrick Waters, Angela Vincent, Eun-Suk Kang, Sujin Lee, Dong Kyoo Lee, Kwang Ho Lee, Byoung Joon Kim
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  ABSTRACT: Neuromyelitis optica (NMO) is an inflammatory demyelinating disorder that predominantly affects the optic nerve and spinal cord; however, symptomatic brain involvement is not rare and is sometimes an initial manifestation in NMO. In this study, we investigated the characteristic features of patients with NMO with symptomatic brain involvement as the initial manifestation of disease (NMObrain) compared with patients with NMO who presented initially with optic neuritis or myelitis (NMOON/myelitis). We retrospectively reviewed 27 consecutive Korean patients with NMO with aquaporin-4 antibodies. Patients with NMObrain (n=9) initially presented with intractable hiccup/nausea/vomiting and/or encephalopathy at a younger age than the patients with NMOON/myelitis (n=18) (p<0.01). During the disease course, the patients with NMObrain continued to show more frequent symptomatic involvement of the brain than the 18 patients with NMOON/myelitis (p<0.05). At the final visit, the mean age was also significantly lower in patients with NMObrain than in patients with NMOON/myelitis (p<0.01); however, the Expanded Disability Status Scale scores, used to evaluate disease progression, were not different between the two groups. Our study suggests that patients with NMO who present initially with symptomatic brain involvement may have earlier disease onset and become disabled at a younger age compared to patients with typical NMO. Additional large scale prospective studies are warranted.
  Journal of Clinical Neuroscience 05/2013;
• Article: Low-grade and anaplastic oligodendrogliomas: Differences in tumour microvascular permeability evaluated with dynamic contrast-enhanced magnetic resonance imaging.

  Zhongzheng Jia, Daoying Geng, Ying Liu, Xingrong Chen, Jun Zhang
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  ABSTRACT: This study was designed to quantitatively assess the microvascular permeability of oligodendroglioma using the volume transfer constant (Ktrans) and the volume of the extravascular extracellular space per unit volume of tissue (Ve) with dynamic contrast-enhanced magnetic resonance imaging (DCE-MRI). We aimed to evaluate the effectiveness of Ktrans and Ve in distinguishing between low-grade and anaplastic oligodendroglioma. The maximal values of Ktrans and Ve for 65 patients with oligodendroglioma (27 grade II, 38 grade III) were obtained. Differences in Ktrans and Ve between the two groups were analysed using the Mann-Whitney rank-sum test. Receiver operating characteristic (ROC) curve analyses were performed to determine the cut-off values for the Ktrans and Ve that could differentiate between low-grade and anaplastic oligodendrogliomas. Values for Ktrans and Ve in low-grade oligodendrogliomas were significantly lower than those in anaplastic oligodendrogliomas (p<0.001 and p<0.001, respectively). ROC curve analysis showed that cut-off values of the Ktrans (0.037min(-1)) and Ve (0.079) could be used to distinguish between low-grade and anaplastic oligodendrogliomas in a statistically significant manner. Our results suggest that DCE-MRI can distinguish the differences in microvascular permeability between low-grade and anaplastic oligodendrogliomas.
  Journal of Clinical Neuroscience 05/2013;
• Article: A randomized open-label observational study to compare the efficacy and tolerability between topiramate and valproate in juvenile myoclonic epilepsy.

  Kang Min Park, Sang Ho Kim, Soon Ki Nho, Kyong Jin Shin, Jinse Park, Sam Yeol Ha, Sung Eun Kim
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  ABSTRACT: Juvenile myoclonic epilepsy (JME) is managed with valproate in most patients; however, valproate is an antiepileptic drug that has relatively severe adverse effects, especially in women. We performed a prospective, open-label, randomized observational study for comparison of efficacy and tolerability between topiramate and valproate in patients with JME. The inclusion criteria were patients with newly diagnosed JME or previously diagnosed JME with a history of a poor response or adverse effects to other antiepileptic drugs. The primary endpoint of this study was percentage of patients who were free of myoclonic seizures for 24weeks in the two groups. The frequency and severity of adverse effects were also assessed. Sixteen patients were randomized to topiramate and 17 to valproate. In the topiramate arm, 11 of 16 patients (68.9%) completed 24-week maintenance therapy and seven of the 11 (64%) were seizure-free. In the valproate arm, 16 of 17 patients (94.1%) completed 24-week follow-up and nine of 16 (56%) were seizure-free. The difference (64% topiramate versus 56% valproate) did not reach statistical significance in this study group (p=0.08, Fisher's exact test). However, the severity of adverse effects was significantly different. Only 1 of 10 adverse effects from topiramate was ranked moderate-to-severe (10%), in comparison with severe rankings for 10 of 17 adverse effects from valproate (59%) (p=0.018, Fisher's exact test). In summary, the efficacy of topiramate and valproate was not different, but the severity of adverse effects was favourable for topiramate. Our findings suggest that valproate may be replaced with topiramate, especially for the patients with JME who do not tolerate valproate.
  Journal of Clinical Neuroscience 05/2013;
• Article: Serum paraoxonase and arylesterase activity and oxidative status in patients with multiple sclerosis.

  Aynur Kirbas, Serkan Kirbas, Omer Anlar, Hasan Efe, Adnan Yilmaz
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  ABSTRACT: The aim of this study was to investigate serum paraoxonase and arylesterase activities, and to determine oxidative status via the measurement of total oxidant status (TOS), total antioxidant status (TAS) and the oxidative stress index (OSI) in patients with relapsing-remitting multiple sclerosis (RRMS). Results were compared with data from healthy controls. A total of 60 subjects, including 30 newly diagnosed and untreated patients with RRMS (20 females, 10 males, 18-40years of age) and 30 healthy controls (20 female, 10 male 20-40years of age) were enrolled in this study. The oxidative status of the RRMS patients was measured by TOS, TAS and estimation of the OSI was made by a new automated method. Paraoxonase (PON1) and arylesterase activities were measured spectrophotometrically. TAS levels of RRMS patients were significantly lower than that of controls (p<0.05). TOS levels of RRMS patients were higher than that of controls (p<0.05). PON1 and arylesterase activities of RRMS patients were lower, but not significantly, than those of controls (p>0.05). There was no correlation between serum PON1 activity and OSİ in patients with RRMS (p>0.05). Hypercholesterolemia was not observed in multiple sclerosis patients. In conclusion, although the mechanism underlying the significant reduction of TAS levels of multiple sclerosis patients compared with those of controls is unknown, the results imply that endogenous antioxidants may have been exhausted by increased oxidative stress and we believe that additional antioxidant treatment might be beneficial for these patients.
  Journal of Clinical Neuroscience 05/2013;
• Article: Thrombo-embolic cerebral infarction secondary to giant Lambl's excrescence.

  Teddy Y Wu, Ivor L Gerber, Richard H Roxburgh
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  ABSTRACT: Lambl's excrescences are common fibrinous strands found at the contact margin of cardiac valves. They are referred to as "giant" when multiple strands form a complex. Embolic stroke secondary to these valvular strands has rarely been described in the literature. We present a patient with recurrent thrombo-embolic cerebral infarction secondary to giant Lambl's excrescence successfully treated with anticoagulation.
  Journal of Clinical Neuroscience 05/2013;
• Article: Prevalence of positive syphilis serology and meningovascular neurosyphilis in patients admitted with stroke and TIA from a culturally diverse population (2005-09).

  Dennis J Cordato, Sanja Djekic, Sanjeev R Taneja, Michael Maley, Roy G Beran, Cecilia Cappelen-Smith, Neil C Griffith, Ibrahim Y Hanna, Suzanne J Hodgkinson, John M Worthington, Alan J McDougall
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  ABSTRACT: The study aims were to determine the prevalence of positive syphilis serology and meningovascular neurosyphilis (NS) in patients admitted with transient ischaemic attack (TIA) and stroke to a tertiary hospital serving a culturally diverse community. A retrospective cohort analysis was conducted using routinely collected administrative data and medical records to identify patients admitted with TIA, stroke and other conditions, with positive syphilis serology, between 2005 and 2009. Direct medical record review confirmed diagnoses of meningovascular NS. Syphilis serology was requested in 27% (893/3270) of all patients with TIA and stroke (2005-09) of whom 4% (38/893) were positive. Thirty-seven patients with positive serology had clinical characteristics consistent with meningovascular NS. Their mean age was 72±13years; 65% were male and 68% had a recorded place of birth in South-East Asia or the Pacific Islands. One of 12 patients with suspected meningovascular NS with cerebrospinal fluid (CSF) analysis had a positive CSF Venereal Disease Research Laboratory (VDRL) test. Three patients (8%) met diagnostic criteria for "definite or probable" meningovascular NS. All three patients with a "definite or probable" meningovascular NS and 15 (44%) of the remainder who had positive serology without confirmation of NS were treated with intravenous or intramuscular penicillin. Lumbar puncture (LP) and penicillin were underutilised in patients with TIA and stroke with positive serology. In conclusion, syphilis testing should be considered part of the diagnostic work-up of TIA and stroke, particularly in ethnically diverse populations. In patients with TIA and stroke with positive syphilis serology, it would seem appropriate to further pursue diagnosis and treatment and in patients unable to undergo LP, empiric treatment for NS should be considered.
  Journal of Clinical Neuroscience 05/2013;
• Article: Clinical characteristics and risk factors of chronic subdural haematoma associated with clipping of unruptured cerebral aneurysms.

  Joji Inamasu, Takeya Watabe, Tsukasa Ganaha, Yasuhiro Yamada, Shunsuke Nakae, Tatsuo Ohmi, Shuei Imizu, Takafumi Kaito, Keisuke Ito, Yuya Nishiyama, Takuro Hayashi, Hirotoshi Sano, Yoko Kato, Yuichi Hirose
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  ABSTRACT: Chronic subdural haematoma (CSDH) is an uncommon but potentially serious complication of clipping unruptured cerebral aneurysms. We conducted a study to identify the patients who are at risk of developing postoperative CSDH. The data from 713 consecutive patients who underwent clipping of unruptured anterior circulation aneurysms were reviewed, and risk factors correlated with CSDH were identified by multivariate regression analysis of demographic variables. Fifteen patients (2.1%) developed CSDH after the surgery. Advanced age (odds ratio [OR] 1.151, 95% confidence interval [CI] 1.051-1.261) and male gender (OR 3.167, 95% CI 1.028-9.751) were correlated with CSDH. Subsequently, all 713 patients were quadrichotomized on the basis of gender and age, with 70years as the cut-off value for age. The frequency of CSDH in men <70years of age was 1.3% and that in men ⩾70years of age was 15.1%, with risk of CSDH was significantly higher in the older men (OR 13.39; 95% CI: 3.42-52.44). The frequency of CSDH in women <70years of age was 0.6% and that in women ⩾70years of age was 3.7%. As in men, the risk of CSDH was significantly higher in the older women (OR 6.69, 95% CI 1.10-40.73). The interval between the aneurysm clipping and CSDH development was 0.5-6months, suggesting that clinical observation should be continued up to 6months after surgery. Although prognosis for patients with a postoperative CSDH complication is generally favourable, the risk of CSDH should be taken into account when considering elective clipping of unruptured aneurysms in patients ⩾70years of age.
  Journal of Clinical Neuroscience 05/2013;
• Article: Intranasal myiasis: A rare complication of transnasal skull base surgery.

  Taha A Jan, Navid Redjal, Brian P Walcott, Sameer A Sheth
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  ABSTRACT: A 57-year-old woman with a history of atypical intracranial meningioma had undergone multiple craniotomies and endoscopic skull base procedures over several years. She presented most recently with nasal discharge consisting of intranasal larvae. Isolated organisms from the nasal cavity and maxillary sinus were identified as blow fly larvae (Calliphoridae family). The patient was treated with transnasal debridement and antibiotic therapy. The organisms were successfully eradicated and she is free from further signs of infection. Intranasal myiasis is an unusual complication of anterior skull base surgery.
  Journal of Clinical Neuroscience 05/2013;
• Article: Vestibular schwannoma mimicking horizontal cupulolithiasis.

  Rachael L Taylor, Luke Chen, Corinna Lechner, Swee T Aw, Miriam S Welgampola
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  ABSTRACT: Positional vertigo and nystagmus can be due to canalithiasis, cupulolithiasis and less commonly, an underlying peripheral or central vestibular disorder. We present a patient with vestibular schwannoma who initially sought treatment for positioning vertigo. Video-oculography on the roll-test revealed direction-changing horizontal apogeotropic nystagmus, consistent with horizontal cupulolithiasis. However, further audio-vestibular investigations and imaging confirmed a right vestibulopathy attributable to a schwannoma of the right vestibular nerve. This case report suggests that vestibular schwannoma should be considered as another potential mimicker of horizontal cupulolithiasis.
  Journal of Clinical Neuroscience 05/2013;
• Article: Intracranial hemorrhage due to intracranial hypertension caused by the superior vena cava syndrome.

  Jiri Bartek, Golbarg Abedi-Valugerdi, Jan Liska, Harriet Nyström, Morten Andresen, Tiit Mathiesen
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  ABSTRACT: We report a patient with intracranial hemorrhage secondary to venous hypertension as a result of a giant aortic pseudoaneurysm that compressed the superior vena cava and caused obstruction of the venous return from the brain. To our knowledge, this is the first patient reported to have an intracranial hemorrhage secondary to a superior vena cava syndrome. The condition appears to be caused by a reversible transient rise in intracranial pressure, as a result of compression of the venous return from the brain. Treatment consisted of surgery for the aortic pseudoaneurysm, which led to normalization of the intracranial pressure and resorption of the intracranial hemorrhage.
  Journal of Clinical Neuroscience 05/2013;
• Article: Superficial siderosis of the central nervous system due to chronic hemorrhage from a giant invasive prolactinoma.

  Jacob Steinberg, José E Cohen, John M Gomori, Shifra Fraifeld, Samuel Moscovici, Guy Rosenthal, Yigal Shoshan, Eyal Itshayek
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  ABSTRACT: Superficial siderosis of the central nervous system (CNS) is a rare disorder caused by deposition of hemosiderin in neuronal tissue in the subpial layer of the CNS due to slow subarachnoid or intraventricular hemorrhage. The most common neurologic manifestations include progressive gait ataxia, sensorineural hearing loss, and corticospinal tract signs. We present a case of superficial siderosis in a 43-year-old man who presented to the Emergency Department with sudden onset bilateral visual deterioration and a loss of consciousness. A hemorrhagic giant prolactinoma was diagnosed based on brain CT scan, T1-weighted MRI, and an endocrine blood examination. Susceptibility-weighted non-contrast MRI showed pathognomonic signs of superficial siderosis in the form of a hypointensity rim surrounding the brainstem, cerebellar fissures, and cranial nerves VII and VIII. This report demonstrates that superficial siderosis can be caused by pituitary apoplexy.
  Journal of Clinical Neuroscience 05/2013;
• Article: Short-lasting unilateral neuralgiform headache with conjunctival injection and tearing caused by a pituitary adenoma.

  T D Musuka, R H Edis, A G Kermode
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  ABSTRACT: Short-lasting unilateral neuralgiform headache with conjunctival injection and tearing (SUNCT) syndrome is a rare primary headache syndrome first described in 1978. We report on a 43-year-old man with a 10 year history of SUNCT in whom a pituitary macroadenoma was eventually detected. His pain rapidly improved with medical treatment of the prolactinoma and we propose that this is a case of symptomatic SUNCT.
  Journal of Clinical Neuroscience 05/2013;
• Article: Hyperdense middle cerebral artery sign is associated with increased risk of hemorrhagic transformation after intravenous thrombolysis for patients with acute ischaemic stroke.

  Ming Zou, Leonid Churilov, Anna He, Bruce Campbell, Stephen M Davis, Bernard Yan
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  ABSTRACT: Haemorrhagic transformation (HT) is an infrequent but serious complication of intravenous thrombolysis therapy (IVT) for acute ischemic stroke. The hyperdense middle cerebral artery sign (HMCAS) is a possible radiological predictor. We aimed to assess the association between HMCAS and HT in a retrospective study. We included all patients with acute anterior circulation ischaemic stroke who received IVT between October 2007 and December 2011. Baseline characteristics were collected, including demographics, stroke risk factors and stroke type. Presence of HMCAS on baseline CT scans was evaluated. Follow-up CT scans were examined for HT, categorised according to the European Australasian Acute Stroke Study (ECASS) classification. The presence of symptomatic intracerebral haemorrhage (sICH) was defined according to Safe Implementation of Thrombolysis in Stroke Monitoring Study (SITS-MOST) criteria. The association between HT and HMCAS was assessed by univariate and multivariate logistic regression analysis. We included 182 consecutive patients treated with IVT in this study. HMCAS was present in 70 patients (38.5%). Patients with HMCAS had higher baseline National Institutes of Health Stroke Scale scores (p<0.001) and more frequent early ischaemic changes on baseline CT scan (p<0.001) than those without HMCAS. We identified 49 instances (26.9%) of HT in 182 follow-up CT scans. HMCAS was associated with HT in univariate analysis (unadjusted odds ratio [OR]=4.151, 95% confidence interval [CI]: 2.081-8.279, p<0.001) and remained an independent risk factor of HT in multivariate analysis (adjusted OR=2.691, 95% CI: 1.231-5.882, p=0.013). There was no statistically significant difference in the frequency of sICH between the HMCAS group and the non-HMCAS group. We concluded that HMCAS is common in anterior circulation infarction and is independently predictive of HT after thrombolytic therapy.
  Journal of Clinical Neuroscience 05/2013;
• Article: Intravenous levetiracetam for treatment of neonatal seizures.

  Abhijeet Rakshasbhuvankar, Shripada Rao, Rolland Kohan, Karen Simmer, Lakshmi Nagarajan
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  ABSTRACT: In this case series we report on eight neonates with refractory seizures who received intravenous levetiracetam when seizures did not respond to two or more conventional anticonvulsants. Six of the eight neonates had an excellent response with either cessation, or reduction in seizures by at least 80%. One neonate showed a partial response while one did not have any reduction in seizure frequency. We did not encounter any adverse effects that could be attributable to levetiracetam.
  Journal of Clinical Neuroscience 05/2013;
• Article: Melkersson-Rosenthal syndrome: a review of seven patients.

  Ruozhuo Liu, Shengyuan Yu
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  ABSTRACT: Melkersson-Rosenthal syndrome (MRS) is a rare disorder consisting of a triad of persistent or recurrent orofacial edema, relapsing facial paralysis and fissured tongue. It is difficult to diagnose and treat. A retrospective review of our MRS patients was performed. The medical files, and treatment, radiologic and histopathologic records of these patients were reviewed. The study group consisted of seven male MRS patients. In four patients, the three classical manifestations were present simultaneously. Neuroimaging examinations found facial soft-tissue thickening and swelling in two patients. The indicators of the disease in our patients were as follows: two had family histories, two had elevated levels of protein in the cerebrospinal fluid, one had an increased immunoglobulin G level in the blood and one had reactive arthritis. All patients responded to systemic corticosteroid treatment. We discuss the genetic, infectious and immunologic factors in the etiology of MRS. Neuroimaging examinations were useful in determining the diagnosis and in determining whether the initial treatment of MRS should be with systemic corticosteroids.
  Journal of Clinical Neuroscience 05/2013;
• Article: Distant Harrington rod migration 35 years after implantation.

  Robert K Lark, Adam M Caputo, Christopher R Brown, Keith W Michael, Julie K Thacker, William J Richardson
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  ABSTRACT: Harrington rods have been successfully implanted in thousands of patients for the correction of scoliotic deformity since the 1950s. An exceedingly rare complication of Harrington rod placement is loosening with resultant migration. The authors present a 50-year-old woman who had a single Harrington rod placed when she was 15years old. Thirty-five years later, she presented with acute sensory changes in her lower extremities. Imaging revealed rod failure and migration of the hardware distally, resulting in penetration of the wall of the rectum. Due to the unique anatomical position of the migrated hardware, sigmoidoscopy was used to directly visualize and remove the rod. The patient ultimately made a full recovery. Rod migration is an exceedingly rare complication that has been described only a few times since the introduction of Harrington rods over 60years ago. The case herein is particularly unique given the extensive period of time that passed before migration (35years) and the use of sigmoidoscopy for hardware removal.
  Journal of Clinical Neuroscience 05/2013;
• Article: Kleine-Levin syndrome with brain atrophy.

  Yu-Ting Shi, Bei-Sha Tang, Hong Jiang
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  ABSTRACT: Kleine-Levin syndrome (KLS) is commonly described as a self-limiting disorder exhibiting episodes of hypersomnia and psychiatric symptoms, but without any enduring disabilities. Recently, reports have shown that persistent or even progressive memory deficits can also be associated with the disorder. Nevertheless, little has been reported about cognitive disturbances in KLS. Our report describes a rare patient with KLS and prominent brain atrophy. A 24-year-old woman developed severe neurological and psychiatric features 12 years previously, 2weeks after she was hit in the head. Although she has had no recurrence of the primary KLS symptoms, she continues to have a cognitive disorder, verbal disability, and whole brain atrophy.
  Journal of Clinical Neuroscience 05/2013;
• Article: A further patient with parasitic myositis due to Haycocknema perplexum, a rare entity.

  Penelope McKelvie, Katrina Reardon, Katherine Bond, David M Spratt, Andrew Gangell, Jane Zochling, John Daffy
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  ABSTRACT: A new genus of nematode, Haycocknema perplexum, causing polymyositis in humans, was first described in two Australian patients from Tasmania in 1998. Three patients with myositis due to the same nematode were reported from northern Queensland in 2008. We report the sixth case from Australia, a 50-year-old man, also from Tasmania. He had a 2-year history of progressive weakness, weight loss of 10kg and dysphagia. Muscle biopsy was initially interpreted as polymyositis with eosinophils. Maximum creatine kinase (CK) level was 5700U/L and full blood examination was normal. He deteriorated after several months of treatment with prednisolone and methotrexate and review of the muscle biopsy showed intramyofibre parasites of H. perplexum. After 3months of treatment with albendazole therapy, he made a very good clinical recovery and his CK decreased to 470U/L. This uniquely Australian parasite can mimic polymyositis and leads to significant irreversible morbidity (two of the previous patients still have weakness and elevated CK after years) and even mortality (one died), if diagnosed late or after corticosteroids. Diagnosis can only be made by histopathology of muscle biopsy.
  Journal of Clinical Neuroscience 05/2013;
• Article: Central nervous system vasculitis secondary to systemic sclerosis.

  Michael S Abers, Emitseilu K Iluonakhamhe, Corey E Goldsmith, Joseph S Kass
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  ABSTRACT: Systemic sclerosis (SSc) or scleroderma is a connective tissue disease with a diverse array of clinical manifestations secondary to underlying fibrosis and autoimmunity. Central nervous system (CNS) impairment is uncommon in SSc. Here we report the fourth known patient with CNS vasculitis caused by SSc. In each previous report, the patient was a middle-aged to elderly female. Our patient was 24years old at the time of presentation, significantly younger than the other reported patients. Importantly, our patient's rapidly progressive clinical course and poor response to immunosuppression have not been reported in patients with CNS vasculitis secondary to scleroderma. Although CNS vasculitis is extremely rare in SSc, our report suggests that clinicians should consider this diagnosis in the differential of SSc patients with neurologic impairment.
  Journal of Clinical Neuroscience 05/2013;
• Article: Leptomeningeal metastasis of an intradural malignant peripheral nerve sheath tumor.

  Andreas M Stark, H Maximilian Mehdorn
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  ABSTRACT: Malignant peripheral nerve sheath tumors (MPNST) are defined as any malignant tumor arising from or differentiating towards the peripheral nerve sheath. Intradural MPNST metastases are very rare. We report, to our knowledge, the first case of leptomeningeal metastasis of a MPNST to the spine and intracranial space. A 56-year-old woman with primary intradural MPNST of the S1 nerve root developed leptomeningeal metastases as well as brain metastases 19months after diagnosis. The patient had a history of non-Hodgkins lymphoma for which she had received irradiation to the spine 15years prior to this presentation. She had no stigmata of neurofibromatosis type 1. Patients with MPNST may also develop leptomeningeal metastases as demonstrated in this patient with intradural post-radiation MPNST.
  Journal of Clinical Neuroscience 05/2013;