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1/30. Intravascular lymphomatosis. A report of ten patients with central nervous system involvement and a review of the disease process.

    The clinical, radiographic, and pathological findings in ten cases of intravascular lymphomatosis with central nervous system involvement seen at our institution over a 15-year period are presented. Nine patients presented with a subacute, progressive multifocal neurologic disorder. Most patients had fever, anemia, and elevation of the erythrocyte sedimentation rate. As the illness evolved, computerized tomography scanning and magnetic resonance imaging showed evidence of multifocal central nervous system disease. angiography was nondiagnostic but suggested vasculitis in six cases. A response to empiric corticosteroid treatment was typical but usually transient. In six patients, the diagnosis was made antemortem by brain biopsy. The prognosis of patients was primarily dependent on early diagnosis and treatment, before massive central nervous system damage occurred. Treatment with chemotherapy, with or without radiotherapy, was associated with stabilization of the disease in three of five patients.
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2/30. Cognitive and behavioral abnormalities in a case of central nervous system whipple disease.

    BACKGROUND: whipple disease is a rare condition characterized by migratory polyarthralgias, fever, and chronic diarrhea. A subset of patients with the disease may either initially have or eventually develop symptoms of central nervous system involvement. DESIGN AND methods: The cognitive and behavioral functioning of a patient with central nervous system involvement from whipple disease was studied during a 7-month period. Serial neuropsychological evaluations were used to quantify the nature of his cognitive and behavioral profile. SETTING: neurology department of a university medical center. RESULTS: A variety of cognitive impairments were noted, most prominently in the domains of sustained attention, memory, executive function, and constructional praxis. There were striking behavioral manifestations as well, including disinhibition and confabulation. CONCLUSIONS: The case demonstrates a degree of higher-order central nervous system dysfunction rarely observed and quantified in connection with whipple disease, and with important implications for differential diagnosis of certain neurologic conditions. We also call attention to some of the neuroanatomical correlates of this encephalopathic condition.
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3/30. Neuro-Behcet's syndrome in a patient not fulfilling criteria for Behcet's disease: clinical features and value of brain imaging.

    central nervous system involvement is rarely an initial presenting manifestation of Behcet's disease (BD). We report the case of a 33-year-old man with recurrent attacks of fever, oral mucosal ulcers, deep venous thrombosis, diplopia, vertigo and headache. Sequential brain magnetic resonance imaging (MRI) scans showed fluctuating lesions of the brain stem, mesencephalon and thalamus. F-18-fluoro-2-deoxy-D-glucose positron emission tomography (FDG-PET) revealed hypometabolism at the parieto-occipital cortex at both sides and the brain stem. Treatment with prednisone and cyclosporine A led to a complete remission and normalisation of MRI and FDG-PET lesions. The present case illustrates the difficulty in the differential diagnosis of early neuro-BD.
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4/30. Cerebrospinal trypanosomiasis masquerading as pulmonary infectious disease in a 1-year-old boy.

    A 1-year-old boy with cerebrospinal trypanosomiasis presented with severe respiratory symptoms, hepatosplenomegaly and no neurological signs of trypanosomiasis. Agitation and high fever on the 2nd day in hospital prompted a lumbar puncture and trypanosomes were recovered from the cerebrospinal fluid.
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5/30. Intracranial meningeal involvement in churg-strauss syndrome.

    We describe the case of a 54-year-old woman with a clinical diagnosis of churg-strauss syndrome (CSS). The patient had a fever of unknown origin, severe headache, progressing left ophthalmoplegia, and visual acuity disturbance. MR imaging revealed diffuse and thick hypointense lesions on T2-weighted images in the frontal meninges and anterior falx cerebri with diffuse enhancement. Similar lesions were also detected in the left superior ophthalmic fissure to the cavernous sinus. Nodular lesions in the fourth ventricle, which might have been the cause of hydrocephalus, were hypointense on T2-weighted images. These MR imaging findings suggested remote granulomatous involvement in the meninges and choroid plexus associated with CSS. To our knowledge, remote meningeal involvement in association with CSS has not been previously reported.
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6/30. west nile virus encephalitis causing fatal CNS toxicity after hematopoietic stem cell transplantation.

    We describe here a patient who died of progressive CNS deterioration following allogeneic stem cell transplant with west nile virus as the sole pathogen on the cerebrospinal fluid and brain tissue analysis. A 50-year-old male with philadelphia chromosome-positive acute lymphocytic leukemia (ALL) underwent allogeneic PBSCT from his HLA identical sister. After engraftment, the patient developed fever with progressive and ultimately fatal neurological deterioration. Imaging studies of the brain including CT and MRI scans were remarkable for mild low attenuation lesions of the white matter. CSF analysis was negative for neoplastic cells, bacteria, AFB, CMV, HSV, fungal infections and leukemic relapse. However, serological analysis of both the serum and CSF was positive for west nile virus-specific IgM antibodies. At autopsy, west nile virus PCR and cultures were positive in the mid-brain tissue. Electron micrographs showed evidence of viral particles. Given the recent increase in the spread of west nile virus infections and the increased susceptibility of BMT patients to infectious complications, west nile virus encephalitis should be considered in patients undergoing transplantation.
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7/30. CNS disease as the main manifestation of hemophagocytic lymphohistiocytosis in two children.

    Hemophagocytic lymphohistiocytosis is a rare and fatal disorder of early infancy, which affects predominantly the mononuclear phagocyte system and is characterized by the presence of fever, hepatosplenomegaly and cytopenia. Neurological symptoms can be extremely variable, ranging from irritability, and convulsions to focal neurological signs. They often develop during disease progression, but can also be the leading initial symptoms. early diagnosis is mandatory, because new treatments, including bone marrow transplantation, appear to be promising. Here we present the clinical, neuroradiological and histopathological findings from two children with progressive CNS disease as the main clinical manifestation of hemophagocytic lymphohistiocytosis. Both children died and diagnosis was only obtained in retrospect after careful review of the histopathological material.
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8/30. polyarteritis nodosa with central nervous system involvement mimicking meningoencephalitis.

    OBJECTIVE: To describe a patient who had polyarteritis nodosa with central nervous system involvement mimicking infectious meningoencephalitis. DESIGN: Case report. SETTING: Pediatric intensive care unit of a university hospital. Patient: A 9-yr-old boy with prolonged fever, headache, decreased level of consciousness, neck stiffness, and papilledema. RESULTS: cerebrospinal fluid examination showed pleocytosis and a high protein level. After neurologic deterioration resulted from the initial treatment with antibiotic, the combination of clinical and laboratory findings with neuroradiologic features led to suspected systemic vasculitis. The patient was treated subsequently with corticosteroid, which resulted in great improvement. biopsy of a skin lesion confirmed the diagnosis of polyarteritis nodosa. CONCLUSIONS: critical care physicians must recognize neurologic manifestation patterns of systemic vasculitides because appropriate diagnosis and therapy result in significantly improved morbidity and mortality.
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9/30. Calgary experience with west nile virus neurological syndrome during the late summer of 2003.

    BACKGROUND: Between August 25 and September 25, 2003 seven patients with west nile virus neurological manifestations were identified through the hospital neurology consultation services in Calgary, alberta, canada. Three of the seven patients were treated with interferon alpha-2b (IFN alpha-2b). In this report we document the clinical characteristics of these seven cases. methods: Clinical and laboratory information was obtained from a retrospective review of patient hospital and clinic charts. patients were included if they had serological evidence of west nile virus infection and had clinical evidence of aseptic meningitis, encephalomyelitis, cerebellar syndrome or motor neuronopathy. Three patients received a treatment course of three million units IFN alpha-2b, administered by subcutaneous injection once per day for 14 days. RESULTS: Four patients had cerebellar signs without change in consciousness, two had both encephalitis and neuromuscular weakness, and one patient had focal lower motor neuron arm weakness. The mean age was 52 (range 24 - 73). All patients had flu-like illness and fever as presenting symptoms and six had severe headaches. Two patients were immunocompromised prior to infection. Two patients with cerebellar signs (one with opsoclonus-myoclonus) improved spontaneously and exhibited only mild residual deficits on discharge. The other two patients with cerebellar findings developed brainstem involvement, one coinciding with and one subsequent to the cerebellar symptoms. Within one week of treatment with IFN alpha-2b these latter two patients showed marked improvement. One patient with encephalitis and neuromuscular weakness, was treated with IFN alpha-2b and subsequently recovered. INTERPRETATION: In this case review of seven patients, multiple neurological symptoms occurred in each patient and the neurological presentation was varied. Four patients had predominant cerebellar findings and one patient had opsoclonus-myoclonus, not previously reported. The marked improvement in three patients who received IFN alpha-2b raises preliminary optimism towards this potential treatment.
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10/30. Occipital intermittent rhythmic delta activity only following eye closure in atypical CNS Salmonellosis.

    OBJECTIVE: A statement recently published on the base of a large retrospective analysis, report that the occipital intermittent rhythmic delta activity (OIRDA) "is associated with epilepsy but not acute encephalopathy" [Gullapalli and Fountain. J Clin Neurophysiol 2003;20:35-41]. Our aim is to report, the exception from a child with an intermittent fever, in which the finding of an occipital intermittent rhythmic delta activity (OIRDA) following the eye closure in the EEG recording was the first clinical sign addressing to a CNS involvement. methods: To review the record from a five-year-old girl with a normal basal electroencephalogram and OIRDA that only appeared following eye closure. RESULTS: We found OIRDA associated with atypical CNS Salmonellosis. Brain MRI and CSF examination confirmed an acute encephalopathy, which was due to salmonella infection.The only symptoms of the infection were episodes of nightly fever that had lasted for four weeks, sometimes associated with headache and vomiting. Both OIRDA only induced by eye closing and other symptoms disappeared after starting antimicrobial therapy. CONCLUSIONS: OIRDA only following eye closure is a non-specific abnormality and the present findings, based on a single case, merely indicate that intracranial infection is among the possible causes. SIGNIFICANCE: The new clinical association is certainly worth recording, as the presence of this electrophysiological sign may provoke clinicians to then delve further into a diagnostic work up.
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