Cases reported "Hydrocephalus"

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1/10. Transient oedema of the cervical spinal cord.

    Transient but very intense oedema of the cervical spinal cord was observed in two patients with obstruction of the cerebrospinal fluid (CSF) pathways. Both presented with hydrocephalus, one due to an infratentorial obstructing mass and the other due to postmeningitic adhesive obstruction of the outlet foramina of the fourth ventricle. In animal experiments with obstruction of CSF pathways (due to outlet foramina obstruction or to downward tentorial herniation) flattening and stretching of the ependymal cells along the central canal is observed, followed by disruption and splitting of the ependymal lining and then by extracellular oedema of the subependymal tissue. Without treatment, frank cavity formation develops in a fourth stage. In our two patients, however, most probably because of appropriate decompressive therapy, the oedema disappeared completely without a residual spinal cord lesion.
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2/10. Use of acetazolamide to decrease cerebrospinal fluid production in chronically ventilated patients with ventriculopleural shunts.

    acetazolamide (ACTZ), a carbonic anhydrase inhibitor, has been shown to decrease cerebrospinal fluid (CSF) production in both in vivo and in vitro animal models. We report two children with hydrocephalus who experienced multiple shunt failures, and who had externalised ventriculostomy drains (EVD) prior to ventriculopleural shunt placement. The effects of increasing doses of ACTZ on CSF production and subsequent tolerance to ventriculopleural shunts were evaluated. The patients had a 48% and a 39% decrease in their EVD CSF output when compared to baseline with maximum ACTZ dose of 75 mg/kg/day and 50 mg/kg/day, respectively (p < 0.05). This is the first report of change in CSF volume in children after extended treatment with ACTZ. ACTZ treatment in mechanically ventilated paediatric patients with hydrocephalus may improve tolerance of ventriculopleural shunts and minimise respiratory compromise. potassium and bicarbonate supplements are required to correct metabolic disturbances.
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3/10. Aqueductal stenosis 9 years after mumps meningoencephalitis: treatment by endoscopic third ventriculostomy.

    INTRODUCTION: hydrocephalus due to aqueductal stenosis following mumps meningoencephalitis is a rare condition, reported only in 16 cases in the literature. The pathogenetic role of the mumps virus in inducing aqueductal stenosis has been demonstrated experimentally in animal models and clinically proven in a few cases. Although obstructive in nature, the post-infectious etiology raises the question as to whether third ventriculostomy is the appropriate treatment. PATIENT: We report a case of hydrocephalus due to pure aqueductal stenosis occurring in an 11-year-old boy who suffered from mumps meningoencephalitis 9 years previously. The boy was successfully treated by endoscopic third ventriculostomy. CONCLUSIONS: The present case offers further evidence of the purely obstructive nature of the hydrocephalus induced by paramyxovirus meningoencephalitis, even if it did occur several years after the infectious disease. The pathogenesis of mumps-induced hydrocephalus and the rationale of treatment are discussed, and the literature is reviewed.
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4/10. brain macrophages and microglia in human fetal hydrocephalus.

    Whereas several studies have addressed the activation of microglia (the resident mononuclear phagocytes of the brain) and macrophages within the nervous system in experimental animal models of congenital and induced hydrocephalus, little is known of their state of activation or regional distribution in human fetal hydrocephalus. This investigation aimed to address such questions. Ten human fetal cases [20-36 gestational weeks (GW) at postmortem] previously diagnosed with hydrocephalus on ultrasound examination in utero, and 10 non-hydrocephalic controls (22-38 GW at postmortem) were assessed immufcnohistochemically with antibodies directed against MHC class II and CD68 antigens, and lectin histochemistry with lycopersicon esculentum (tomato lectin). Adjacent sections were also immunoreacted with an antiserum to laminin to detect cerebral blood vessels. Eight out of the 10 hydrocephalus cases showed numerous CD68 and tomato lectin-positive macrophages located at focal regions along the ependymal lining of the lateral ventricles (particularly within the occipital horn). However, only five of these cases demonstrated MHC class II positive macrophages associated with the ventricular lining. Microglial reactivity within periventricular regions could also be identified using the lectin in four cases, two of which were also immunoreactive with CD68 (but not with MHC class II). By comparison, in control cases five out of 10 fetal brains (aged between 20 and 24 GW) showed few or no ependymal or supraependymal macrophages. One case at 28 GW, and cases at 32 and 38 GW (two of which were diagnosed with intrauterine hypoxic-ischemia) did, however, show some MHC class II (CD68 negative) cells located at the ependymal surface. Nevertheless, these were not as numerous or intensely immunoreactive as in the hydrocephalus cases. microglia interspersed throughout the intermediate zone and circumscribing the basal ganglia were within normal confines in all cases examined. Hydrocephalic cases additionally showed focal regions of hypovascularization or alterations in the structure and orientation of capillaries within periventricular areas, compared to controls. The macrophage response detected at the ependymal lining of the ventricles and within the periventricular area in hydrocephalus may be related both to the severity of hydrocephalus and the age of the fetus.
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5/10. Hypersexuality following septal injury.

    Hypersexuality is an uncommon consequence of brain injury. We report two cases of markedly increased sexual activity following septal damage sustained in the course of placement of ventriculoperitoneal shunts. These two cases, observations in animal experiments, and descriptions of altered behavior in humans with localized brain dysfunction indicate that a circuit involving the septal nuclei has an important role in the mediation of sexual behavior.
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6/10. Limb reduction defects in humans associated with prenatal isotretinoin exposure.

    Retinoic acid has long been used to induce limb reductions defects in experimental animal studies. No limb malformations, however, have been reported among malformed retinoic acid-exposed human fetuses from case reports or epidemiologic studies. We report a child and a fetus with limb reduction malformations following maternal use of isotretinoin (13-cis-retinoic acid) during the first trimester of pregnancy. The child had a unilaterally absent clavicle and nearly absent scapula, with a short humerus and short, synostotic forearm bones. He also had ventriculomegaly and developmental delay, minor dysmorphic facial features, and a short sternum with a sterno-umbilical raphe. The fetus had a unilaterally absent thumb with normal proximal bony structures. Other findings included hydrocephalus, craniofacial anomalies, thymic agenesis, supracristal ventricular septal defect, single umbilical artery, anal and vaginal atresia, and urethral agenesis with dysplastic, multicystic kidneys. Although the limb malformations were quite dissimilar, a number of anomalies that are frequently found among isotretinoin-exposed fetuses/infants were present in both cases. This increases the probability that retinoic acid caused these limb defects, but a causal association cannot be conclusively drawn on the basis of these two retrospective case reports.
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7/10. Obstructive hydrocephalus following Pantopaque myelography.

    A case with delayed development of obstructive hydrocephalus most probably caused by Pantopaque myelography is reported. At autopsy it was found that the foramina of Magendi and Luschka were occluded by granulation tissue in which the most common findings were multinucleated giant cells containing calcareous and unstained vacuoles, frequently surrounding empty hollows, many lymphocytes and plasma cells. As compared with two other similar cases and several animal investigations this shows that the reaction of the tissue is typical of a Pantopaque reaction.
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8/10. Cerebral abscess caused by cladosporium bantianum. Case report.

    A case is presented of a cerebral abscess in a 59-yr-old woman caused by the dematiaceous fungus cladosporium bantianum (Sacc.) Borelli (= cladosporium trichoides Emmons). The morphological and physiological properties of the isolate and its pathogenicity for experimental animals were investigated and compared with some other cladosporium species. The neurotropism exhibited by C. bantianum in human infections was found also in the test animals. Despite surgical excision and therapy with the antifungal drugs amphotericin b and flucytosine, the case had a fatal outcome, the patient dying from a pulmonary embolism 6 mth after the onset of symptoms. The clinical entity of central nervous system infections by dematiaceous fungi is discussed in relation to the present case.
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9/10. Cerebroarthrodigital syndrome: a newly recognized formal genesis syndrome in three patients with apparent arthromyodysplasia and sacral agenesis, brain malformation and digital hypoplasia.

    We describe three patients with a complex syndrome of apparent arthromyodysplasia, dyscephaly, sacral agenesis, and hypoplastic digitis. Cause is unknown, but an environmental cause is suspected on the basis of ergotamine exposure in one case and diazoxide intake in another, together with suggestive similarities to anomalies seen in animals treated with these drugs and to calves with the Australian hydranencephaly/arthrogryposis syndrome caused by Akebane or Aino virus. Pathogenetically the primary defect may be a neural tube-neural crest dysplasia with multiple secondary and tertiary manifestations and deformities.
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10/10. Functional obstruction of an antisiphon device by raised tissue capsule pressure.

    The proper function of an antisiphon device (ASD) requires that it be exposed externally to atmospheric pressure and that its mobile membrane be free to move. A 4-year-old boy who had an ASD placed for postshunt subdural hematomas presented with functional obstruction of the ASD-symptomatic ventriculomegaly despite evidence of patency of the shunt system by radionuclide flow study. At surgery, the distal shunt system including the ASD was infused with normal saline before and after surgical exposure of the device. The pressures required to maintain a flow rate of 5 ml/h and 50 ml/h were 27 mm Hg and 30 mm Hg, respectively. After surgical exposure, these pressures fell to 0 mm Hg and 5 mm Hg, respectively, for the same infusion rates. Surgical exposure removes the effects of the tissue capsule, including the overlying skin and a collagenous sheath, restoring the external pressure to atmospheric pressure. Functional obstruction of ASDs occurs because of raised ambient pressure from the tissue capsule acting to depress the mobile membrane of the ASD, increasing its resistance to flow. This case confirms previously reported effects of subcutaneous implantation of ASDs in experimental animals and is the probable explanation for reported functional obstruction of ASDs in other patients.
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