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Increased tau burden in the cortices of progressive supranuclear palsy presenting with corticobasal syndrome.

The objective of this study is to better define the pathological characteristics of pathologically proven progressive supranuclear palsy (PSP) presenting with the corticobasal syndrome (CBS). PSP is characterized by early falls, vertical supranuclear ophthalmoplegia, and axial rigidity, whereas asymmetric limb features, including rigidity, bradykinesia, apraxia, alien limb phenomena, and cortical sensory loss are characteristic of CBS. We investigated clinicopathological characteristics of 5 cases of PSP that presented with CBS (CBS-PSP). Comprehensive pathological analysis was undertaken to determine the presence of concomitant pathological processes as well as quantitative tau burden in cortical regions of CBS-PSP, compared with 8 typical PSP cases (Typ-PSP). The clinical features in the CBS-PSP cases included asymmetrical features, apraxia, alien limb phenomena, and progressive aphasia. All cases had Parkinsonism, and vertical supranuclear ophthalmoplegia was noted in all but 1 case of CBS-PSP. Secondary neuropathological diagnoses included argyrophilic grain disease (AGD) in 1 of the 8 cases of Typ-PSP, whereas Alzheimer's disease (AD), Lewy body disease, AGD, and vascular disease was found in 3 cases of CBS-PSP. Image analysis of cortical tau burden performed in 8 Typ-PSP and 3 CBS-PSP cases revealed a significant increased tau burden in mid-frontal and inferior-parietal cortices in the CBS-PSP cases. This study demonstrates that when PSP presents as CBS, it is most likely due to either a concurrent cortical pathology from a secondary process such as AD or from the primary pathology of PSP extending into cortical areas that are primarily and commonly affected in CBD.

Aged↗

Extracranial internal carotid artery dissections: noniatrogenic traumatic lesions.

Noniatrogenic traumatic extracranial internal carotid artery dissections were encountered in six patients (five men and one woman) 31 to 62 years old. All but one had overt cerebral ischemia manifest by paresis (three), sensory deficits (three), aphasia (three), or amaurosis fugax (two), One patient had an asymptomatic carotid artery bruit. Cerebral arteriography established the diagnosis in all cases. Internal carotid artery impingement between the mandible and transverse processes of the second and third cervical vertebrae, or undue stretching over these vertebral structures, were the most likely primary mechanisms of injury. Secondary complications, a result of distal thromboembolism, were evident in two patients. Direct cerebral revascularization, staged internal carotid artery constriction and ligation, as well as intensive nonoperative therapy were valid therapeutic options. There were no deaths. Treatment relieved transient ischemic symptoms or arrested progression of established neurologic deficits in each case. In select patients, early surgical intervention may lessen attending neurologic sequelae.

Adult↗

[Frontal dementia or dementia praecox? A case report of a psychotic disorder with a severe decline].

INTRODUCTION: Many authors have described these last years the difficulty to establish a differential diagnosis between schizophrenia and frontotemporal dementia. However treatment and prognosis of these two separate diseases are not the same. Schizophrenia is a chronic syndrome with an early onset during teenage or young adulthood period and the major features consist of delirious ideas, hallucinations and psychic dissociation. However a large variety of different symptoms describes the disease and creates a heterogeneous entity. The diagnosis, exclusively defined by clinical signs, is then difficult and has led to the research of specific symptoms. These involve multiple psychological processes, such as perception (hallucinations), reality testing (delusions), thought processes (loose associations), feeling (flatness, inappropriate affect), behaviour (catatonia, disorganization), attention, concentration, motivation (avolition), and judgement. The characteristic symptoms of schizophrenia have often been conceptualised as falling into three broad categories including positive (hallucination, delision), negative (affective flattening, alogia, avolition) and disorganised (poor attention, disorganised speech and behaviour) symptoms. No single symptom is pathogonomonic of schizophrenia. These psychological and behavioural characteristics are associated with a variety of impairments in occupational or social functioning. Cognition impairments are also associated with schizophrenia. Since the original clinical description by Kraepelin and Bleuler, abnormalities in attentional, associative and volitional cognitive processes have been considered central features of schizophrenia. Long term memory deficits, attentional and executive dysfunctions are described in the neurocognitive profile of schizophrenic patients, with a large degree of severity. The pathophysiology of schizophrenia is not well known but may be better understood by neuronal dysfunctions rather than by a specific anatomical abnormality. Frontotemporal lobar degeneration (FTLD) is one of the most common causes of cortical dementia. FTLD is associated with an anatomical atrophy that can be generalised, with a frontotemporal or focal lobar predominance. Histologically there is severe neuronal loss, gliosis and a state of spongiosis. In a minority of case Pick cells and Pick bodies are also found. The usual clinical features of FTLD are divided in three prototypic syndromes: frontotemporal dementia (FTD), progressive non-fluent aphasia (PA) and semantic dementia (SD). FTD is the most common clinical manifestation of FTLD. FTD is first characterised by profound alteration in personality and social conduct, characterised by inertia and loss of volition or social disinhibition and distractibility. There is emotional blunting and loss of insight. Speech output is typically economical, leading ultimately to mutism, although a press of speech may be present in some overactive, disinhibited patients. Memory is relatively preserved in the early stage of the disease. Cognitive deficits occur in the domains of attention, planning and problems solving, whereas primary tools of language, perception and spatial functions are well preserved. PA is an initial disorder of expressive language, characterised by effortful speech production, phonologic and grammatical errors. Difficulties in reading and writing also occur but understanding of word meaning is relatively well preserved. In SD a severe naming and word comprehension impairment occur on the beginning in the context of fluent, effortless, and grammatical speech output. There is also an inability to recognise the meaning of visual percepts. The clinical syndromes of FTLD are associated with the brain topography of the degeneration. So considerable clinical overlap can exist between schizophrenia and FTLD and the object of the following case report is to remind the difficulty to make a differential diagnosis between these two pathologies. CASE REPORT: A 34 year old non-married man is admitted in mental health district of a general hospital for behavioural disturbances that include repeated aggressions towards his family. At initial interview visual and auditives hallucinations are described. The patient doesn't care about these abnormalities and a poverty of speech is observed. The affects, globally blunted, show some degree of sadness however. The patient's birth and early development were unremarkable. At the age of 26, the patient dismissed from his job because of poor performance and absenteeism. He spent a lot of time watching TV, showed poverty of speech and become sometimes angry and violent without an explanation. He was hospitalised for several months and a schizophrenia including predominant negative features, hallucinations and delusion was diagnosed. He was treated with bromperidol, could go back to home and was followed by a general practitioner for 8 years. The patient had a stereotyped way of life during these years with a poor communication and little activity. During the months preceding the current hospitalisation, these characteristics and avolition emphasised, urinary incontinence appeared. The patient receives risperidone 8 mg/day associated with citalopram 40 mg/day during several months of hospitalisation. No significant evolution is observed regarding apathic and stereotyped way of live. The capacity of communication remains very poor. Neurocognitive assessments reveal multiple and severe dysfunctions. Memory, executive and attentional tasks are extremely disturbed. Physical and neurological examinations reveal an isolated bilateral Babinski sign. Cerebral scanner and magnetic resonance show bifrontal atrophy and PET scan is normal. There are no significant abnormalities found on blood and urine samples and on lumbar puncture. The patient is sent to a chronic neuropsychiatric hospital and the treatment is stopped. One year later, a comparative evaluation is realised. The general clinical state shows no evolution. Neurocognitive assessments are repeated and severe dysfunctions are observed with more perseverations. DISCUSSION: A diagnosis of FTLD for this patient can be discussed regarding clinical features, neurocognitive testings and neuroradiological findings. Schizophrenia is a major differential diagnosis. Psychotic symptoms like hallucinations and age of onset are essential observations for the diagnosis of schizophrenia but can not exclude FTLD. Memory, intellectual functions, executive and attentional abilities may all be disturbed in schizophrenia and FTLD. Cerebral abnormalities well established in schizophrenia are lateral ventricles enlargements. Frontal lobar atrophy is a major argument for FTLD and is only a sporadic finding in schizophrenic populations. Schizophrenia and FTLD could be comorbid diseases by several ways. CONCLUSION: A differential diagnosis between schizophrenia and FTLD is difficult to establish. Schizophrenia is a heterogeneous disease with a large variety of cognitive dysfunctions. Neurocognitive tools may improve our knowledge of schizophrenia.

Adult↗

Intra-arterial cisplatin for the treatment of malignant gliomas.

Cisplatin (DDP) is a chemotherapeutic agent that has shown efficacy against primary CNS malignancies. Intra-arterial (IA) administration of DDP to patients with brain tumors should produce higher peak levels of drug than intravenous (IV) administration of an identical dose and reduce systemic toxicity. Twelve patients with malignant glioma were entered into the study. All had failed irradiation, 11 had failed IA BCNU. Each patient received IA DDP, 58-100 mg/m2, into the internal carotid artery at four to six week intervals. One of 12 patients had a partial response of 6 months. The remaining 11 patients had progressive disease or severe complications. Toxicity included seizures in four patients, weakness and/or aphasia in four patients, coma in two patients, and visual deterioration in two patients. IA DDP has very limited efficacy in patients with malignant gliomas after failure of nitrosoureas and is associated with an unacceptable level of toxicity. IA DDP may be more effective when used as initial chemotherapy of malignant gliomas.

Adolescent↗

[Severe neurological and obstetrical complications in a patient with antiphospholipid syndrome].

Antiphospholipd syndrome (APS) is a disease characterised by venous and arterial thrombosis or recurrent foetal loss, which are associated with antiphospholipid antibodies or/and lupus anticoagulant. Clinical symptoms ofAPS are assocciated with presence of noninflammatory thrombosis ocluding arteries or venes. Symptoms of APS are often very dramatic what can be illustrated by the presented case of 40 year patient. At the age of 25 after a miscarriage the patient developed tetraparesis and motor aphasia in the course of thrombosis in central nervous system vessels. In physical examination besides neurological symptoms reticular livedo was found on the trunk and limbs. Serological tests have revealed presence of high titre of IgG and IgM anticardiolipin antibodies and lupus anticoagulant (LA). CT examination revealed hypodensic foci in left parietal lobe, and abnormal EEG findings were observed in fronto-temporal leads of left hemisphera of brain. At that point the patient did not meet the criteria of connective tissue diseases, including lupus. The diagnosis of primary APS was suggested. The patient received anti-aggregation treatment and also immunosuppressive drugs (azathioprine and prednison) due to progression of neurological manifestations. 3 years later, the second pregnancy ended in the 27th week with intrauterine fetal death. During the 3rd pregnancy, 2 years afterwards, the patient was treated with heparin, aspirin and intravenous immunoglobulin. The pregnancy finished with a successful delivery at term, the newborn was in good condition. During the following pregnancy the symptoms of preeclampsia occurred at 36/37th week but the newborn was delivered in a good condition after a caesarean section. At the age of 36 patient developed ischemic brain stroke with left-side hemiparesis inspite of anti-aggregation and immunosupressive (prednison and azathioprine) treatment. At that time homogenic type antinuclear antibodies (ANA) 1:2560, anti-beta2-glycoprotein antibodies (beta2-GPI), aCL antibodies in medium titer and thrombocytopenia were found. The patient was treated with heparin, cyclophosphamide and methyloprednisolon intravenously. During rehabilitation process gradual improvement of cognitive functions, speech and motorical functions was observed. Recently high titre of ANA and recurent thrombocytopenia < 100,000/mm3 were present. Maybe during the further follow-up, 14 years after the first symptoms of APS, the patient will develop full blown symptoms of SLE.

Anti-Inflammatory Agents↗

[Anterior type dementia from the viewpoint of neuropathology].

Anterior type dementia is characterized by primary degeneration in the anterior brain region and represents pick symptom. Anterior type dementia includes several pathological disease entities, almost of them usually exhibit variation of atrophic brain regions in greater or lesser degree. This variation of each disease cause confusion, namely, the same clinical manifestations occur even if it is a different disease and diversely, different clinical manifestations can occur even if it is the same disease entity. This is the reason why discrepancy between clinical diagnosis and pathology diagnosis occurs sometimes in anterior type dementia. For reasonable clinical diagnosis, anterior type dementia, separate from neuropathological diseases entity, should be deal with clinical symptom-complex, which consists of frontotemporal dementia (FTD) progressive nonfluent aphasia (PA) and semantic dementia (SD). FTD, PA and SD have, though broadly, corresponding responsible brain lesion respectively.

Atrophy↗

Dementia in human populations exposed to neurotoxic agents: a portable microcomputerized dementia screening battery.

Dementia has been described as a clinical syndrome associated with chronic diffuse cerebral hemispheric dysfunction resulting in multiple involvement of cognition, memory, language, visual-spatial skills, and personality. While the specific diseases causing dementia are diverse, the severity of the dementia is directly correlated with the loss of functional brain tissue, independent of the primary neuropathology. Many neurotoxicants are pharmacologically nonselective. Thus, chronic exposure to these agents would be expected to result in progressive, diffuse impairment of CNS functioning that would present clinically as a substance-induced dementia. This suggests that diagnostic techniques developed for the early detection of incipient dementia in the aged might prove useful in screening for dementia in younger populations. A microcomputer based Psychometric Assessment System (PAS) and Dementia Screening Battery (DSB) with high predictive value for differentiating the normal age-related changes in cognition from those associated with incipient dementia in the aged have been developed. The DSB is DSM-III compatible as it evaluates intellectual deterioration, malignant memory loss, amnestic aphasia, and spatial disorientation. A diagnosis of dementia is obtained by findings of intellectual deterioration and malignant memory loss and either amnestic aphasia or spatial deterioration.

Aphasia↗

Phase II trial of intracarotid BCNU and cisplatin in primary malignant brain tumors.

Intracarotid BCNU (100 mg/m2) and cisplatin (60 mg/m2) were administered to 36 patients with malignant brain tumors recurrent or progressive after cranial irradiation. Courses of therapy were repeated at 4-6 week intervals. Of 23 evaluable patients with recurrent glioma, 9 (39%) had tumor regression by CT scan and 3 had stable disease. The median time to tumor progression for responding patients was 37 weeks. For all patients with primary tumors it was 14 weeks. Six of 9 patients with no prior chemotherapy had a response and 1 had stable disease. Of 14 patients who had received prior chemotherapy, 3 had a response and 2 had stable disease. Survival ranged from 9 weeks to 95+ weeks (median 34 weeks) from start of therapy. Six of 23 patients with primary tumors are alive 1 year or more following therapy. Four of 11 patients with brain metastases had a response and 2 had stable disease. Major neurologic toxicity of intracarotid BCNU and cisplatin appeared cumulative and consisted of reversible hemiparesis in 3% of 118 courses, TIA in 1%, expressive aphasia in 9%, lethargy in 3%, seizures in 12%, and reversible confusion in 1%. Retinal toxicity consisted of mild blurring of vision in 4 patients and ipsilateral blindness in 5 patients. Three of 22 patients who had received supraophthalmic infusion later developed evidence of leukoencephalopathy. Intracarotid BCNU and cisplatin appears to have modest increase in activity over intracarotid cisplatin alone (Cancer 54:794, 1984), however, neurologic and retinal toxicity may also be increased.

Adult↗

Frontotemporal lobar degeneration: a consensus on clinical diagnostic criteria.

OBJECTIVE: To improve clinical recognition and provide research diagnostic criteria for three clinical syndromes associated with frontotemporal lobar degeneration. METHODS: Consensus criteria for the three prototypic syndromes-frontotemporal dementia, progressive nonfluent aphasia, and semantic dementia-were developed by members of an international workshop on frontotemporal lobar degeneration. These criteria build on earlier published clinical diagnostic guidelines for frontotemporal dementia produced by some of the workshop members. RESULTS: The consensus criteria specify core and supportive features for each of the three prototypic clinical syndromes and provide broad inclusion and exclusion criteria for the generic entity of frontotemporal lobar degeneration. The criteria are presented in lists, and operational definitions for features are provided in the text. CONCLUSIONS: The criteria ought to provide the foundation for research work into the neuropsychology, neuropathology, genetics, molecular biology, and epidemiology of these important clinical disorders that account for a substantial proportion of cases of primary degenerative dementia occurring before the age of 65 years.

Dementia↗

Progress in hereditary tauopathies: a mutation in the Tau gene (G389R) causes a Pick disease-like syndrome.

We describe the clinical and pathologic phenotypes of the G389R mutation in exon 13 of the Tau gene. Progressive aphasia and memory disturbance are the initial signs and begin in the fourth or fifth decade of life, followed by apathy, indifference, hyperphagia, rigidity, pyramidal signs and dementia. Death occurs after two to five years. Magnetic resonance imaging and neuropathologic studies show frontal and temporal atrophy. Pick body-like and axonal filamentous inclusions found in the neocortex and subcortical white matter, respectively, are tau immunoreactive. Immunoblot analysis of sarkosyl-insoluble tau shows two major bands of 60 and 64 kDa that, upon dephosphorylation, resolve into four bands of three- and four-repeat isoforms. Isolated tau filaments are often straight and occasionally twisted. Recombinant mutant tau protein shows a reduced ability to promote microtubule assembly, suggesting that this may be the primary effect of the mutation. The present findings indicate that the G389R mutation in Tau can cause a dementia similar to that in Pick's disease.

Adult↗

Moyamoya and Down syndrome. Clinical and radiological features.

BACKGROUND AND PURPOSE: Moyamoya disease is a chronic occlusive cerebrovascular disorder characterized by progressive stenosis of the supraclinoid internal carotid artery, with the secondary development of enlarged basal collateral vessels. It may occur as a primary disease or as a syndrome in association with a variety of conditions, and its pathogenesis remains unexplained. There are relatively few reports describing the occurrence of moyamoya in Down syndrome. The aim of this study is to describe the clinical and radiological features of moyamoya syndrome associated with Down syndrome (MM-DS) and to explore theories of moyamoya pathogenesis in these patients. METHODS: Seven children with MM-DS underwent brain imaging, transfemoral angiography, and serial neurological exams. Neurological deficits, poststroke recovery, radiographic infarct characteristics, and angiographic abnormalities were reviewed. RESULTS: The clinical and radiological features of primary moyamoya disease overlap with those of MM-DS. Hemiplegia and aphasia were the most common presentations. Motor recovery was excellent in five of seven cases. Cerebral infarcts were superficial or deep and can occur in a watershed distribution. Angiography demonstrated involvement of the internal carotid artery and its branches bilaterally in all seven cases and the posterior cerebral arteries in four cases. CONCLUSIONS: The clinical and radiological features of MM-DS overlap with primary moyamoya disease. We postulate that a protein encoded on chromosome 21 may be related to the pathogenesis of moyamoya disease. Although the neuronal substrate is abnormal in Down syndrome patients, recovery from hemiplegic stroke in patients with MM-DS is comparable to recovery in patients with primary moyamoya.

Adolescent↗

Frontal lobe degeneration of non-Alzheimer type.

In a longitudinal prospective study of dementias, several hundred cases have been examined from a clinical, brain imaging, neurochemical and neuropathological point of view. Frontal lobe degeneration of non-Alzheimer type (FLD) was the second most common primary degenerative dementia found in about 10% of the material. FLD has a consistent pathology and a characteristic clinical picture, which have been described by several independent research groups. The cortical degeneration mainly involves frontal or frontotemporal grey matter, without the circumscribed or knife-blade atrophy seen in Pick's disease. The degeneration involves predominantly frontal areas, including the insula and cingulate gyrus in its anterior parts. The striate body is normal or only slightly altered. The pathological changes are non-specific, with neuronal loss, slight gliosis and spongiosis but none or few senile plaques, tangles, congophilic vessels or Pick cells. Pathological changes are in some respects similar to those in amyotrophic lateral sclerosis. FLD is a slowly progressive dementia with personality changes, lack of insight, disinhibition, stereotypy and later apathy. There is also progressive dynamic aphasia which ends in mutism and amimia. Memory, spatial ability and receptive language functions are comparatively spared. Psychotic symptoms, emotional reactions, hypochondriasis and a Klüver-Bucy-like syndrome are sometimes observed. Electroencephalography is normal, at least during the early stage, while functional brain imaging such as regional cerebral blood flow reflects the frontal pathology. It is possible to achieve early diagnosis and differentiation from Alzheimer's disease and cerebrovascular dementia by clinical examination with neuropsychological assessment supported by brain imaging, and in the future probably various biological markers. The aetiology is unknown but there is a positive family history for dementia of similar type in about 50% of post-mortem verified cases.

Cerebrovascular Circulation↗

Tau gene mutation G389R causes a tauopathy with abundant pick body-like inclusions and axonal deposits.

Exonic and intronic mutations in Tau cause familial neurodegenerative syndromes characterized by frontotemporal dementia and dysfunction of multiple cortical and subcortical circuits. Here we describe a G389R mutation in exon 13 of Tau. When 38 years old, the proband presented with progressive aphasia and memory disturbance, followed by apathy, indifference, and hyperphagia. Repeated magnetic resonance imaging showed the dramatic progression of cerebral atrophy. Positron emission tomography revealed marked glucose hypometabolism that was most severe in left frontal, temporal, and parietal cortical regions. Rigidity, pyramidal signs and profound dementia progressed until death at 43 years of age. A paternal uncle, who had died at 43 years of age, had presented with similar symptoms. The proband's brain showed numerous tau-immunoreactive Pick body-like inclusions in the neocortex and the fascia dentata of the hippocampus. In addition, large numbers of tau-positive filamentous inclusions were present in axons in the frontal, temporal, and parietal lobes. Immunoblot analysis of sarkosyl-insoluble tau showed 2 major bands of 60 and 64 kDa. Upon dephosphorylation, these bands resolved into 4 bands consisting of three- and four-repeat tau isoforms. Most isolated tau filaments were straight and resembled filaments found in Alzheimer disease and some frontotemporal dementias with tau mutations. A smaller number of twisted filaments was also observed. Biochemically, recombinant tau proteins with the G389R mutation showed a reduced ability to promote microtubule assembly, suggesting that this may be the primary effect of the mutation. Taken together, the present findings indicate that the G389R mutation in Tau can cause a dementing condition that closely resembles Pick's disease.

Adult↗

Language functions in Huntington's disease.

A comprehensive language test battery (Aachen Aphasia Test) was administered to 45 patients in the early, middle or later stages of Huntington's disease (HD) and to 20 control subjects. In spontaneous speech, many HD patients exhibited a loss of conversational initiative. Dysarthria was a common finding. Reading skills were found to be impaired mainly as a consequence of dysarthria; some HD patients displayed visual dyslexia. In addition to the characteristic disturbances of writing skills due to the choreiform movement disorder, the writing of HD patients with advanced dementia indicated constructional dysgraphia, characterized by frequent omissions, perseverations and substitutions. HD patients exhibited no evidence of word-finding difficulty or other semantic deficits in spontaneous speech. There was, however, a marked impairment in visual confrontation naming, with a significant rise in naming error rate as the disease progressed in severity. In most instances, the inappropriate names referred to an object visually similar to the target object, suggesting that visual misperception is the major cause of the naming disorder in HD. Syntactical structure of spontaneous speech was typically reduced to short, simple sentence construction. Verbal stereotypes were only rarely encountered and occurred late in the course of the disease. Tests of language comprehension reflected the general degree of dementia. It is concluded that there are no primary language changes in HD. Instead, a variety of language impairments develop secondary to other neurological and neuropsychological changes.

Adult↗

Perceptual and linguistic factors in the language impairment of developmental dysphasics: an experimental investigation with the token test.

Twelve developmental dysphasics and twelve control children were tested on De Renzi and Vignolo's Token Test. The performance of the dysphasics, though significantly poorer than that of the controls on Parts 2-5 of the test, seemed to follow a similar pattern. Both groups were errorless on the initial parts of the test. As the demand on auditory retention increased on subsequent parts of the test, the performance of both groups progressively deteriorated. However, the addition of grammatical complexity on Part 5, rather than causing even further deterioration in performance, actually served to improve the performance of both groups, though not significantly. Although the pattern of errors made by the two groups was similar, the quantity of errors made by the dysphasics, as compared to the normal control children, demonstrated the magnitude of the dysphasic children's receptive language impairment. Analysis of errors indicated that dysphasic children have greater difficulty with auditory retention and verbal memory than they do with the grammatical or structural complexity of language. It is suggested that the observed gross language impairment of developmentally dysphasic children does not result, at least primarily, from a specific inability to analyze the linguistic components of language. Rather, the language impairment of these children appears to reflect their primary inability to analyze the rapid stream of acoustic information which characterizes speech and is essential to normal speech perception and language development.

Aphasia↗

Involved-field radiotherapy and intra-Ommaya methotrexate/cytarabine in patients with AIDS-related lymphomatous meningitis.

PURPOSE: To evaluate combined limited-field radiotherapy and concentration times time (C X T) intra-CSF chemotherapy in patients with AIDS-related lymphomatous meningitis (LM). PATIENTS AND METHODS: Fourteen men and one woman with AIDS had cytologically documented LM. Eleven patients had systemic non-Hodgkin's lymphoma (NHL) (all B-cell histology, including six immunoblastic, four large cell, one small cell) with leptomeningeal metastases and four patients had primary CNS lymphoma (PCNSL) (all B-cell histology, including two immunoblastic, two large cell) with CSF dissemination. Presenting neurologic examinations included cranial neuropathies (n = 7), normal (n = 4), abulia (n = 2), paraparesis (n = 2), ataxia (n = 1), hemiparesis (n = 1), and aphasia (n = 1). Standardized pretreatment evaluations included contrast cranial magnetic resonance/computed tomography (MR/CT), placement of an intraventricular reservoir, CT myelogram/contrast spine MR, ophthalmologic examination, and indium 111-pentetic acid (DTPA) CSF flow studies. Regions of bulky or symptomatic disease were treated with limited-field radiation therapy, which included whole brain in 10 patients combined with spinal cord irradiation in five patients. Concurrent systemic chemotherapy was administered in 12 patients. All patients were scheduled to receive intraventricular methotrexate (MTX) 2 mg/d for 5 consecutive days biweekly for 8 weeks (induction), followed in cytologically responding patients by MTX administered in a similar manner every 4 weeks (maintenance). In MTX-responsive and consenting patients with cytologic relapse, intraventricular cytarabine (ara-C) was administered, 25 mg/d for 3 consecutive days weekly for 4 weeks (induction), followed by ara-C administered in a similar manner every 4 weeks (maintenance). CSF cytology and neurologic examinations were performed biweekly. RESULTS: In 13 assessable patients (two patients refused CNS directed therapy following standardized pretreatment evaluations), median time to tumor progression was 60 days (range, 3 to 260) and median survival duration was 125 days (range, 44 to 260). Response rate, determined clinically (four of 13 patients) and cytologically (nine of 13), was 69%. Complications included reservoir infection (n = 2) and myelosuppression (n = 11); the latter was felt to be a consequence of coadministered systemic chemotherapy. CONCLUSION: There were no treatment-related deaths. We conclude that involved-field irradiation and intraventricular MTX/ara-C is effective palliative treatment of AIDS-related LM.

Adult↗

[Where fronto-temporal dementia should be placed in the history of Pick's disease and related disorders].

Pick syndrome, non-Alzheimer type dementia with primary degeneration in the anterior portion of the brain, has been shown to include several disease-entities besides classic Pick's disease. Fronto-temporal dementia (FTD) proposed by Lund & Manchester group is a concept that includes Pick's disease, and its "clinical diagnostic features" are useful because they distinguish dementia with frontal and anterior temporal involvement from Alzheimer type dementia. FTD is classified into three types, the frontal lobe degeneration (FLD) type, Pick type and motor neuron disease (MND) type, and neuropathological diagnostic features of each type are presented. In Japan, however, cases neuropathologically corresponding to the FLD type, which has mild frontal dominant degeneration with heavy heredity, have not been previously reported. Classic Japanese Pick's disease is consistent with Pick type, however, cases of Pick's disease with and without Pick body exist in nearly the same number in Japan. The latter may include heterogeneous types, which should be elucidated. MND type corresponds to Japanese cases, called Yuasa-Mitsuyama disease or dementia with motor neuron disease, and could be actually diagnosed because such patients do not exhibit severe personality change and accompany symptoms of amyotrophic lateral sclerosis or spinal progressive muscular atrophy in their clinical course. In addition, Pick syndrome includes progressive subcortical gliosis, corticobasal degeneration, basophilic inclusion body disease and fronto-temporal dementia with parkinsonism linked chromosome 17. Progressive aphasia and semantic dementia are neuropsychologically important concepts, however, they are symptom-complex and are dominated only by where the degeneration begins and how it progresses. They should not be confused with pathologically confirmed disease-entities.

Dementia↗

[Surgical removal of unilateral vestibular schwannomas after failed Gamma Knife radiosurgery].

One of the primary criticismes of vestibular schwannoma (VS) radiosurgery is that the risk of surgical morbidity is increased for patients whose tumor progresses after the procedures. We reviewed the French experience of operated patients after failed Gamma Knife radiosurgery. From July 1992 to January 2002, 25 out of the 1000 treated patients underwent another treatment procedure for a gamma knife failure. Excluding the NF2 patients, 21 patients have been operated and the present study shows the data collected for 20 of them. In order to analyze the difficulties observed during the surgery, a questionnaire was filled by the surgeons. The mean interval between radiosurgery and removal was 36 Months, from 10 to 83 Months. The mean increase in Volume was 559% (37 to 3036%, median 160%). Evolution of the Koos grading was found from 8 grade II, 10 grade III et 2 grade IV to 10 grade III and 10 grade IV. Patients have been operated for radiological tumor growth in 7 cases and for clinico-radiological evolution in 13 cases. In 9 cases, the surgeon considered that he had to face unusual difficulties mainly because of adhesion of the tumor to neurovascular structures. Tumor removal was total in 14 cases, near total in 4 cases and subtotal in 2 cases. One case of venous infarction was noticed at the second day following surgery responsible of hemiparesis and aphasia that gradually recovered. At last follow-up examination, facial nerve was normal (House and Brackmann grade I and II) in 10 cases while it was a grade III in 7 cases and grade IV and V in 3 cases. We recommend that the decision for surgical removal of growing vestibular schwannoma after Gamma Knife treatment should be done after a sufficiently long follow-up period. Our results show that the quality of removal and of facial nerve preservation might be impaired by radiosurgery in half of cases. However these results do not support a change in our policy of radiosurgical treatment of small to medium size vestibular schwannoma.

Adult↗