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Mahmoud Hamdy Kamel, Chris Lim, Michael Kelleher, Kristian Aquilina, Catherine Keohane and George Kaar

✓ Chordoma is a locally invasive tumor of low metastatic potential. Only six cases of chordoma that metastasized to the brain are found in the English literature. Most of these lesions were clinically silent and all were associated with extraneural metastases. The authors report a case of symptomatic brain metastasis from a sacrococcygeal chordoma in the absence of other metastases. The incidence, sites, and factors predictive of chordoma metastasis are discussed.

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Mahmoud Hamdy Kamel, Martin Murphy, Michael Kelleher, Kristian Aquilina, Chris Lim and Charles Marks

✓ The authors report on a case of schistosomiasis of the spinal cord in an individual returning to Ireland after a 25-year residence in Africa, where the infection affects approximately 200 million people.

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Mahmoud Hamdy Kamel, Michael Kelleher, Kristian Aquilina, Chris Lim, John Caird and George Kaar

Object

Neuroendoscopists often note pulsatility or flabbiness of the floor of the third ventricle during endoscopic third ventriculostomy (ETV) and believe that either is a good indication of the procedure's success. Note, however, that this belief has never been objectively measured or proven in a prospective study. The authors report on a simple test—the hydrostatic test—to assess the mobility of the floor of the third ventricle and confirm adequate ventricular flow. They also analyzed the relationship between a mobile floor (a positive hydrostatic test) and prospective success of ETV.

Methods

During a period of 3 years between July 2001 and July 2004, 30 ETVs for obstructive hydrocephalus were performed in 22 male and eight female patients. Once the stoma had been created, the irrigating Ringer lactate solution was set at a 30-cm height from the external auditory meatus, and the irrigation valve was opened while the other ports on the endoscope were closed. The ventricular floor ballooned downward and stabilized. The irrigation valve was then closed and ports of the endoscope were opened. The magnitude of the upward displacement of the floor was then assessed. Funneling of the stoma was deemed to be a good indicator of floor mobility, adequate flow, and a positive hydrostatic test. All endoscopic procedures were recorded using digital video and recordings were subsequently assessed separately by two blinded experienced neuroendoscopists. Patients underwent prospective clinical follow up during a mean period of 11.2 months (range 1 month–3 years), computerized tomography and/or magnetic resonance imaging studies of the brain, and measurements of cerebrospinal fluid pressure through a ventricular reservoir when present. Failure of ETV was defined as the subsequent need for shunt implantation. The overall success rate of the ETV was 70% and varied from 86.9% in patients with a mobile stoma and a positive hydrostatic test to only 14.2% in patients with a poorly mobile floor and a negative test (p < 0.05). The positive predictive value of the hydrostatic test was 86.9%, negative predictive value 85.7%, sensitivity 95.2%, and specificity 66.6%.

Conclusions

The authors concluded that the hydrostatic test is an easy, brief test. A positive test result confirms a mobile ventricular floor and adequate flow through the created ventriculostomy. Mobility of the stoma is an important predictor of ETV success provided that there is no obstruction at the level of the arachnoid granulations or venous outflow. A thin, redundant, mobile third ventricle floor indicates a longstanding pressure differential between the third ventricle and the basal cisterns, which is a crucial factor for ETV success. A positive hydrostatic test may avert the need to insert a ventricular reservoir, thus avoiding associated risks of infection.

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Kristian Aquilina, Christopher Lim, Mahmoud Hamdy Kamel, Charles J. Marks, Michael G. O'Sullivan and Catherine Keohane

✓ Epithelioid hemangioendothelioma (EH) is a rare tumor of vascular origin. The authors describe two cases of spinal EH, one involving the T-10 vertebra and the second involving the upper cervical spine. In the first case the patient underwent resection of the tumor; this case represents the longest reported follow-up period for spinal EH. In the second case, extensive involvement of C-2, C-3, and C-4 as well as encasement of both vertebral arteries precluded safe tumor resection, and posterior occipitocervical stabilization was performed. The patient subsequently died of metastatic disease. The findings in these two cases underscore the difficulty in predicting the clinical behavior of spinal EH based solely on histological and clinical features as well as the uncertainty of the roles of surgery, chemotherapy, and radiotherapy in the oncological management of a spinal tumor for which clinical data are very limited.

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Mahmoud Hamdy Kamel, Michael Kelleher, Kristian Aquilina, Chris Lim, John Caird and George Kaar

Object. Neuroendoscopists often note pulsatility or flabbiness of the floor of the third ventricle during endoscopic third ventriculostomy (ETV) and believe that either is a good indication of the procedure's success. Note, however, that this belief has never been objectively measured or proven in a prospective study. The authors report on a simple test—the hydrostatic test—to assess the mobility of the floor of the third ventricle and confirm adequate ventricular flow. They also analyzed the relationship between a mobile floor (a positive hydrostatic test) and prospective success of ETV.

Methods. During a period of 3 years between July 2001 and July 2004, 30 ETVs for obstructive hydrocephalus were performed in 22 male and eight female patients. Once the stoma had been created, the irrigating Ringer lactate solution was set at a 30-cm height from the external auditory meatus, and the irrigation valve was opened while the other ports on the endoscope were closed. The ventricular floor ballooned downward and stabilized. The irrigation valve was then closed and ports of the endoscope were opened. The magnitude of the upward displacement of the floor was then assessed. Funneling of the stoma was deemed to be a good indicator of floor mobility, adequate flow, and a positive hydrostatic test. All endoscopic procedures were recorded using digital video and recordings were subsequently assessed separately by two blinded experienced neuroendoscopists. Patients underwent prospective clinical follow up during a mean period of 11.2 months (range 1 month–3 years), computerized tomography and/or magnetic resonance imaging studies of the brain, and measurements of cerebrospinal fluid pressure through a ventricular reservoir when present. Failure of ETV was defined as the subsequent need for shunt implantation. The overall success rate of the ETV was 70% and varied from 86.9% in patients with a mobile stoma and a positive hydrostatic test to only 14.2% in patients with a poorly mobile floor and a negative test (p < 0.05). The positive predictive value of the hydrostatic test was 86.9%, negative predictive value 85.7%, sensitivity 95.2%, and specificity 66.6%.

Conclusions. The authors concluded that the hydrostatic test is an easy, brief test. A positive test result confirms a mobile ventricular floor and adequate flow through the created ventriculostomy. Mobility of the stoma is an important predictor of ETV success provided that there is no obstruction at the level of the arachnoid granulations or venous outflow. A thin, redundant, mobile third ventricle floor indicates a longstanding pressure differential between the third ventricle and the basal cisterns, which is a crucial factor for ETV success. A positive hydrostatic test may avert the need to insert a ventricular reservoir, thus avoiding associated risks of infection.

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Kristian Aquilina, Donncha F. O’Brien, Michael A. Farrell and Ciaran Bolger

✓The authors report on the case of a craniopharyngioma arising in the cerebellopontine angle (CPA) in a patient with Gardner syndrome. Although familial adenomatous polyposis (FAP) is associated with intracranial neoplasms, the current case is only the third reported craniopharyngioma in a patient with Gardner syndrome. Two of these tumors, including that of the current case, originated in the CPA, an unusual location for craniopharyngiomas. The literature concerning FAP and its associations with intracranial neoplasia, as well as the pathogenesis of craniopharyngiomas in the posterior fossa, is discussed.

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Kristian Aquilina, Catherine Hobbs, Shobha Cherian, Alexander Tucker, Helen Porter, Andrew Whitelaw and Marianne Thoresen

Object

The combination of intraventricular hemorrhage (IVH) and posthemorrhagic ventricular dilation (PHVD) remains an important cause of disability in children surviving prematurity. Currently, there is no clear agreement on the management of neonatal IVH, apart from the eventual insertion of a shunt to control PHVD. Cerebrospinal fluid (CSF) shunts are associated with a relatively high complication rate in this population. The development of new treatment options requires greater understanding of the pathophysiological mechanisms of IVH and PHVD, as well as an opportunity to monitor closely their effects on the immature brain. The authors have developed a neonatal large animal model of IVH with long-term survival, allowing the full development of PHVD.

Methods

Fourteen piglets that were 3 to 24 hours old were randomized to receive slow injections of autologous blood, autologous blood with elevated hematocrit, or artificial CSF after induction of general anesthesia. A fourth group served as controls. All animals underwent surgery to form an artificial fontanelle at the bregma. Physiological parameters, including intracranial pressure and electroencephalography, were monitored during injection.

Results

Serial cranial ultrasonography studies performed during the 23- to 44-day survival period demonstrated progressive ventricular dilation in the animals injected with blood. Ventricular volumes, measured with image analysis software, confirmed the highest dilation after injection of blood with an elevated hematocrit. Histological evaluation showed fibrosis in the basal subarachnoid space of hydrocephalic piglets.

Conclusions

This piglet model closely replicates human neonatal IVH and PHVD. It allows detailed physiological and ultrasonographic monitoring over a prolonged survival period. It is suitable for evaluation of noninvasive as well as surgical options in the management of IVH and PHVD.

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Amit Parmar, Kristian Aquilina and Michael R. Carter

Chronic obstructive hydrocephalus is known to cause ventricular diverticula and, rarely, spontaneous ventriculostomy. The authors present the case of a patient in whom a spontaneous third ventriculostomy was identified with long-standing hydrocephalus secondary to aqueductal stenosis. To their knowledge, this is the first report in which a spontaneous stoma in the floor of the third ventricle was evaluated using endoscopy and cerebrospinal fluid dynamics studies. Both studies confirmed that the spontaneous stoma is similar in structure and function to surgical third ventriculostomy.

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Kristian Aquilina, Thomas E. Merchant, Carlos Rodriguez-Galindo, David W. Ellison, Robert A. Sanford and Frederick A. Boop

Malignant transformation of craniopharyngioma is a rare event; only 3 cases have been reported in children, all in the Japanese literature. The authors describe 2 additional pediatric cases. Treatment in both of these cases consisted of multiple resections and external beam radiation therapy (EBRT). Malignant transformation occurred 7 and 8 years after EBRT. The authors also review another 6 cases in adults. A possible causative association with radiation therapy is discussed. As radiation is currently an important option in the management of craniopharyngiomas, this association requires further study.

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Kristian Aquilina, Dave F. Clarke, James W. Wheless and Frederick A. Boop

Temporal lobe encephaloceles can be associated with temporal lobe epilepsy. The authors report on the case of an adolescent with multiple microencephaloceles, in the anterolateral middle fossa floor, identified at surgery (temporal lobectomy) for intractable partial-onset seizures of temporal origin. Magnetic resonance imaging revealed only hippocampal atrophy. Subdural electrodes demonstrated ictal activity arising primarily from the anterior and lateral temporal lobe, close to the microencephaloceles, spreading to the anterior and posterior mesial structures. Pathological examination revealed diffuse temporal gliosis involving the hippocampus, together with microdysgenesis of the amygdala. The literature on epilepsy secondary to encephaloceles is reviewed and the contribution of the microencephaloceles to the seizure disorder in this patient is discussed.