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Surgical treatment of intractable epilepsy associated with focal cortical dysplasia

Roberto Jose Diaz, Elisabeth M. S. Sherman, and Walter J. Hader

Focal cortical dysplasias (FCDs) are congenital malformations of cortical development that are a frequent cause of refractory epilepsy in both children and adults. With advances in structural and functional neuroimaging, these lesions are increasingly being identified as a cause of intractable epilepsy in patients undergoing surgical management for intractable epilepsy. Comprehensive histological classification of FCDs with the establishment of uniform terminology and reproducible pathological features has aided in our understanding of FCDs as an epilepsy substrate. Complete resection of FCDs and the associated epileptogenic zone can result in a good surgical outcome in the majority of patients.

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Surgical management of hypothalamic hamartomas in patients with gelastic epilepsy

Bassam Addas, Elisabeth M. S. Sherman, and Walter J. Hader

Gelastic epilepsy (GE) associated with hypothalamic hamartomas (HHs) is now a well-characterized clinical syndrome consisting of gelastic seizures starting in infancy, medically refractory seizures with or without the development of multiple seizure types, and behavioral and cognitive decline. It has been postulated that the development of the HH-GE syndrome is a result of a progressive epileptic encephalopathy or secondary epileptogenesis, which is potentially reversible with treatment of the HH. A variety of surgical options for the treatment of HHs exist, including open and endoscopic procedures, radiosurgery, interstitial radiotherapy, and stereotactic radiofrequency thermocoagulation. Surgical treatment can result in seizure freedom in up to 50% of patients and can be accompanied by significant improvements in behavior, cognition, and quality of life. Partial treatment of HHs may be sufficient to reduce seizure frequency and improve behavior and quality of life with less risk. A component of reversible cognitive dysfunction may be present in some patients with an HH-GE syndrome.

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Endoscopic third ventriculostomy to treat hydrocephalus associated with macrocephaly-cutis marmorata telangiectatica congenita

Report of 2 cases

Alim P. Mitha, Kelly J. Bullivant, Julie L. Lauzon, and Walter J. Hader

Macrocephaly-cutis marmorata telangiectatica congenita is a rare overgrowth syndrome commonly associated with hydrocephalus. Although the pathophysiological characteristics of the hydrocephalus in this syndrome is not fully known, previous reports have described its treatment with ventriculoperitoneal shunt placement. The authors describe 2 cases of macrocephaly-cutis marmorata telangiectatica congenita successfully treated for progressive hydrocephalus with endoscopic third ventriculostomy. Both patients experienced clinical and radiographic stabilization following treatment, and these findings offer insight into the pathophysiology of the hydrocephalus and its ideal management.

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Death and Third Ventriculostomy

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Application of intraoperative high-field magnetic resonance imaging in pediatric neurosurgery

Clinical article

Ron Levy, Robin G. Cox, Walter J. Hader, Terry Myles, Garnette R. Sutherland, and Mark G. Hamilton

Object

Over the past decade, the use of intraoperative MR (iMR) imaging in the pediatric neurosurgical population has become increasingly accepted as an innovative and important neurosurgical tool. The authors summarize their experience using a mobile 1.5-T iMR imaging unit with integrated neuronavigation with the goal of identifying procedures and/or pathologies in which the application of this technology changed the course of surgery or modified the operative strategy.

Methods

A database has been prospectively maintained for this patient population. The authors reviewed the hospital charts and imaging results for all patients in the database. This review revealed 105 neurosurgical procedures performed in 98 children (49 male and 49 female) between March 1998 and April 2008. Intradissection (ID) and/or quality assurance images were obtained at the discretion of the surgeon.

Results

The median age at surgery was 12 years (4 months–18 years). One hundred intracranial and 5 spinal procedures were performed; 22 of these procedures were performed for recurrent pathology. Surgical planning scans were obtained for 102 procedures, and neuronavigation was used in 93 patients. The greatest impact of iMR imaging was apparent in the 55 procedures to resect neoplastic lesions; ID scans were obtained in 49 of these procedures. Further surgery was performed in 49% of the procedures during which ID scans had been obtained. A smaller proportion of ID scans in the different cranial pathology groups (5 of 21 epilepsy cases, 4 of 9 vascular cases) resulted in further resections to meet the surgical goal of the surgeon. Two ID scans obtained during 5 procedures for the treatment of spinal disease did not lead to any change in surgery. Postoperative scans did not reveal any acute adverse events. There was 1 intraoperative adverse event in which a Greenberg retractor was inadvertently left on during ID scanning but was removed after the scout scans.

Conclusions

The application of iMR imaging in the pediatric neurosurgical population allows, at minimum, the opportunity to perform less invasive surgical exposures. Its potential is greatest when its high-quality imaging ability is coupled with its superior neuronavigation capabilities, which permits tracking of the extent of resection of intracranial tumors and, to a lesser extent, other lesions during the surgical procedure.

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Endoscopic third ventriculostomy for treatment of adult hydrocephalus: long-term follow-up of 163 patients

Albert M. Isaacs, Yarema B. Bezchlibnyk, Heather Yong, Dilip Koshy, Geberth Urbaneja, Walter J. Hader, and Mark G. Hamilton

OBJECTIVE

The efficacy of endoscopic third ventriculostomy (ETV) for the treatment of pediatric hydrocephalus has been extensively reported in the literature. However, ETV-related long-term outcome data are lacking for the adult hydrocephalus population. The objective of the present study was to assess the role of ETV as a primary or secondary treatment for hydrocephalus in adults.

METHODS

The authors performed a retrospective chart review of all adult patients (age ≥ 18 years) with symptomatic hydrocephalus treated with ETV in Calgary, Canada, over a span of 20 years (1994–2014). Patients were dichotomized into a primary or secondary ETV cohort based on whether ETV was the initial treatment modality for the hydrocephalus or if other CSF diversion procedures had been previously attempted respectively. Primary outcomes were subjective patient-reported clinical improvement within 12 weeks of surgery and the need for any CSF diversion procedures after the initial ETV during the span of the study. Categorical and actuarial data analysis was done to compare the outcomes of the primary versus secondary ETV cohorts.

RESULTS

A total of 163 adult patients with symptomatic hydrocephalus treated with ETV were identified and followed over an average of 98.6 months (range 0.1–230.4 months). All patients presented with signs of intracranial hypertension or other neurological symptoms. The primary ETV group consisted of 112 patients, and the secondary ETV consisted of 51 patients who presented with failed ventriculoperitoneal (VP) shunts. After the initial ETV procedure, clinical improvement was reported more frequently by patients in the primary cohort (87%) relative to those in the secondary ETV cohort (65%, p = 0.001). Additionally, patients in the primary ETV group required fewer reoperations (p < 0.001), with cumulative ETV survival time favoring this primary ETV cohort over the course of the follow-up period (p < 0.001). Fifteen patients required repeat ETV, with all but one experiencing successful relief of symptoms. Patients in the secondary ETV cohort also had a higher incidence of complications, with one occurring in 8 patients (16%) compared with 2 in the primary ETV group (2%; p = 0.010), although most complications were minor.

CONCLUSIONS

ETV is an effective long-term treatment for selected adult patients with hydrocephalus. The overall ETV success rate when it was the primary treatment modality for adult hydrocephalus was approximately 87%, and 99% of patients experience symptomatic improvement after 2 ETVs. Patients in whom VP shunt surgery fails prior to an ETV have a 22% relative risk of ETV failure and an almost eightfold complication rate, although mostly minor, when compared with patients who undergo a primary ETV. Most ETV failures occur within the first 7 months of surgery in patients treated with primary ETV, but the time to failure is more prolonged in patients who present with failed previous shunts.

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Cortical dysplastic lesions in children with intractable epilepsy: role of complete resection

Walter J. Hader, Mark Mackay, Hiroshi Otsubo, Shiro Chitoku, Shelly Weiss, Lawrence Becker, O. Carter Snead III, and James T. Rutka

Object. The authors conducted a study to determine seizure-related outcomes in a group of pediatric patients with pathologically proven focal cortical dysplasia (FCD) treated by focal cortical resections and multiple subpial transections (MSTs).

Methods. The authors performed a retrospective review of pediatric patients in whom surgery was conducted to treat medically refractory epilepsy secondary to cortical dysplasia between April 1989 and January 2001. Diagnostic studies included preoperative scalp electroencephalography (EEG), magnetic resonance (MR) imaging, positron emission tomography (PET), and magnetoencephalography (MEG). Intraoperative electrocorticography (ECoG) or extraoperative subdural grid EEG monitoring was performed in all patients. Seizure outcome was classified using the Engel scheme. The authors analyzed nine data points and compared these with seizure outcome, including seizure semiology, MR imaging, PET and MEG data, as well as location of resection, intracranial video-EEG findings, MSTs, postresection ECoG data, and histological findings.

The authors analyzed data obtained in 39 children in whom the follow-up interval after epilepsy surgery was at least 18 months. Patients had suffered epilepsy for a mean of 7.7 years prior to surgical intervention and their mean age at treatment was 9.6 years (range 2 months–18 years). A good seizure-related outcome was demonstrated in 28 patients (72%), including 21 (54%) who were free of seizures (Engel Class I) and seven (18%) in whom seizures were rare (Engel Class II). In 11 patients seizure-related outcome was less favorable, including six (15%) with worthwhile improvement involving some seizures (Engel Class III) and five (13%) with no postoperative seizure improvement (Engel Class IV). There was no significant correlation between seizure outcome and data related to seizure characteristics, MR imaging, PET scanning, MEG, location of resection, intracranial video-EEG, postresection ECoG, and histological findings. Eight (50%) of 16 patients who underwent MSTs in addition to incomplete resection of FCD experienced a good outcome (Engel Class I and II). Twenty (87%) of 23 patients in whom resection of FCD was complete and in whom MSTs were not performed experienced a good seizure outcome (p < 0.05).

Conclusions. Complete resection of FCD results in good seizure outcome in a majority of children. When conducted in conjunction with incomplete cortical resection, MSTs do not improve seizure outcome in patients with FCD. Focal cortical dysplasia located outside of eloquent cortex and complete excision of the lesion are the most important predictors of seizure outcome.

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Death after late failure of third ventriculostomy in children

Report of three cases

Walter J. Hader, James Drake, Douglas Cochrane, Owen Sparrow, Edward S. Johnson, and John Kestle

✓ Late failure following successful third ventriculostomy for obstructive hydrocephalus is rare, and death caused by failure of a previously successful third ventriculostomy has been reported only once. The authors present three patients who died as a result of increased intracranial pressure (ICP) after late failure of a third ventriculostomy.

Through a collaborative effort, three patients were identified who had died following third ventriculostomy at one of the authors' institutions. A 13-year-old girl with neurofibromatosis Type 1 underwent third ventriculostomy for obstructive hydrocephalus caused by a tectal lesion. Three years later her condition deteriorated rapidly over the course of 6 hours and she was found dead at home. A 4-year-old boy treated with third ventriculostomy for aqueductal stenosis presented 2 years postoperatively with symptoms of increased ICP. This patient suffered a cardiorespiratory arrest while under observation and died despite external ventricular drainage. A 10-year-old boy with previous ventriculoperitoneal (VP) shunt placement underwent conversion to a third ventriculostomy and shunt removal. Eight months after the procedure his condition deteriorated, with evidence of raised ICP, and he underwent emergency insertion of another VP shunt, but remained in a vegetative state and died of complications. Neuropathological examinations in two cases demonstrated that the third ventriculostomy was not patent, and there was also evidence of increased ICP.

Late failure of third ventriculostomy resulting in death is a rare complication. Delay in recognition of recurrent ICP symptoms and a false feeling of security on the part of family and caregivers because of the absence of a shunt and the belief that the hydrocephalus has been cured may contribute to fatal complications after third ventriculostomy. Patients with third ventriculostomies should be followed in a manner similar to patients with cerebrospinal fluid shunts.