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      Leptomeningeal dissemination of pilocytic astrocytoma at diagnosis in childhood: two cases report Translated title: Disseminação leptomeníngea de astrocitoma pilocítico ao diagnóstico: relato de dois casos


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          Pilocytic astrocytoma (PA) is a benign tumor that rarely spread along the neuraxis. At the moment there are no more than five cases of leptomeningeal dissemination (LD) from PA at diagnosis described in the literature. Different patterns of presentation or recurrence may be noted: local recurrence, malignant transformation, multicentric disease or metastatic disease. LD and multicentric disease can be distinct pathological entities. We report two cases and analyse literature, emphasizing leptomeningeal spread at presentation. Hydrocephalus, biopsy and parcial ressection are likely to be favorable factors to the occurrence of LD. Otherwise, LD may be part of natural history of PA, as evidenced by its ocurrence in non-treated cases.

          Translated abstract

          Astrocitoma pilocítico (AP) é tumor benigno que raramente se dissemina ao longo do neuroeixo. Até o momento não há mais que cinco casos de AP que se tenham apresentado com disseminação leptomeníngea (DL) descritos na literatura. Diferentes padrões de apresentação ou recorrência podem ser observados: recorrência local, transformação maligna, doença multicêntrica ou doença metastática. DL e doença multicêntrica podem ser entidades diferentes. Relatamos dois casos e analisamos a literatura. Hidrocefalia, biópsia e ressecção parcial são provavelmente fatores predisponentes à DL. Por outro lado, DL pode ser parte da história natural de AP, como pode ser evidenciado pela sua ocorrência em casos não tratados.

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          Most cited references35

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          Dissemination of low grade intracranial astrocytomas in children.

          The authors report three cases of histologically benign intracranial astrocytomas that developed in children and disseminated within the neuraxis. Multicentric disease was evident at the time of diagnosis in two of these patients, one of whom subsequently developed peritoneal seeding of tumor after placement of a ventriculoperitoneal shunt. To our knowledge, this latter represents the first documented case of extraneural seeding of a benign astrocytoma. All three children are currently alive and well 17-117 months after the diagnosis of tumor dissemination. We review the literature regarding this uncommon problem and discuss the pathophysiology and treatment options. Our results indicate that the presence of disseminated disease may not preclude long term survival, provided the lesions are truly benign histologically and that adjuvant therapy is instituted promptly.
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            Low-grade astrocytoma with neuraxis dissemination at diagnosis.

            Little is known about low-grade astrocytoma with neuraxis dissemination at diagnosis. A review of medical records identified this phenomenon in eight of 150 pediatric patients evaluated between 1985 and 1994 for histologically confirmed low-grade astrocytoma. These patients (five male and three female) ranged in age from 5 months to 20 years (median 8 years). Symptoms of neuraxis disease were minimal or absent. Primary tumor sites were the hypothalamus in four cases, brainstem/spinal cord in three, and temporal lobe in one. Patterns of dissemination (evaluated by computerized tomography and/or magnetic resonance imaging techniques) appeared to be related to the primary site: hypothalamic tumors metastasized along the ventricular cerebrospinal fluid pathways, and tumors in other locations disseminated along subarachnoid pathways. Following initial treatment with chemotherapy (in three), partial resection (in one), radiation therapy (in three), and chemotherapy plus irradiation (in one), four patients required salvage therapy for progressive or recurrent disease. Seven of the eight patients are alive with stable or progressive disease 6 to 105 months postdiagnosis (median 15 months). Low-grade astrocytoma with initial neuraxis dissemination is responsive to chemotherapy and radiation, a proportion showing periods of stable disease. The optimum therapy or combination of therapies remains unclear.
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              Value of cerebrospinal fluid cytology for the diagnosis of malignancies in the central nervous system.

              The authors present a retrospective analysis of the results of the cytological examinations of cerebrospinal fluid (CSF) samples and tumor-cyst aspirates deriving from 262 patients treated for malignant intracranial primary and secondary tumors, and vertebral and peridural metastastic processes. Positive preoperative CSF samples were found in 15.3% of all cases of primary cerebral malignancies (13.9% of all gliomas) and positive postoperative CSF samples were found in 40% (91% of the medulloblastoma cases). In all cases of single or multiple secondary cerebral tumors, positive preoperative CSF samples were found in 20%.

                Author and article information

                Arquivos de Neuro-Psiquiatria
                Arq. Neuro-Psiquiatr.
                Academia Brasileira de Neurologia - ABNEURO (São Paulo, SP, Brazil )
                September 2003
                : 61
                : 3B
                : 842-847
                [01] São Paulo SP orgnameSão Paulo University orgdiv1Clinics Hospital orgdiv2School Of Medicine Brazil
                S0004-282X2003000500025 S0004-282X(03)06100325

                This work is licensed under a Creative Commons Attribution 4.0 International License.

                : 12 May 2003
                : 10 February 2003
                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 35, Pages: 6

                SciELO Brazil

                quimioterapia,radioterapia,cerebrospinal fluid,multicentric disease,leptomeningeal dissemination,pilocytic astrocytoma,radiation therapy,metastasis,metástases,líquor,doença multicêntrica,disseminação leptomeníngea,astrocitoma pilocítico,hydrocephalus,chemotherapy


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