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Yu-tze Ng, John F. Kerrigan and Harold L. Rekate

Object

The purpose of this study was to report the cases of five patients with status epilepticus who were treated successfully with neurosurgery.

Methods

Five children presented with refractory status epilepticus, including complex partial status epilepticus, epilepsia partialis continua and “status gelasticus.” Multiple medical therapies had failed to control the seizures, and focal resection was performed. Seizures were fully controlled in four patients, and in one patient seizure frequency was reduced by more than 90%.

Conclusions

Refractory status epilepticus is associated with a high mortality rate and by definition does not respond well to medical treatment, even optimal medical treatment. Neurosurgical treatment should be considered as another therapeutic option once medical management has failed, and neurosurgery should not necessarily be a last resort.

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Yu-tze Ng, John F. Kerrigan, Erin C. Prenger, William L. White and Harold L. Rekate

P The authors report the case of a 12-year-old girl with Pallister—Hall syndrome, long-standing refractory, symptomatic epilepsy, mental retardation, and panhypopituitarism in whom two rare, deep midline lesions were detected. She underwent successful transsphenoidal resection of the Rathke cleft cyst and transcallosal resection of the hypothalamic hamartoma within a 4-day period without complications. Neuropathological studies confirmed the neuroimaging diagnoses for the two lesions. The patient has been seizure free for 6 months postoperatively.

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Iman Feiz-Erfan, Eric M. Horn, Harold L. Rekate, Robert F. Spetzler, Yu-Tze Ng, Jeffrey V. Rosenfeld and John F. Kerrigan III

Object

The authors provide evidence that direct resection of hypothalamic hamartomas (HHs) can improve associated gelastic and nongelastic seizures.

Methods

Ten children younger than 17 years of age underwent resection of HHs (nine sessile and one pedunculated) that were causing refractory epilepsy. Lesions were approached from above transventricularly through a transcallosal anterior interforniceal approach in six cases, endoscopically through the foramen of Monro in one, and from below with a frontotemporal craniotomy including an orbitozygomatic osteotomy in three. Medical charts were reviewed retrospectively, and follow-up data were obtained through office records and phone calls.

Follow-up periods ranged between 12 and 84 months (mean 16.8 months). All patients in whom the approach was from above had sessile HHs. Five were free from seizures at follow up and two had a reduction in seizures of at least 95%. The transventricular route allowed excellent exposure and visualization of the local structures during resection. Among the three patients in whom the approach was from below, one became free of seizure after two procedures and one had a 75% reduction in epilepsy; the latter two had sessile HHs. The exposure was inadequate, and critical tissue borders were not readily apparent. Although the HH was adequately exposed and resected, the epilepsy persisted in the third patient, who had a pedunculated lesion. The overall rate of major permanent hypothalamic complications appeared to be slightly lower for the orbitozygomatic osteotomy group.

Conclusions

Sessile lesions are best approached from above. Approaches from below adequately expose pedunculated hamartomas. The likelihood of curing seizures seems to be higher when lesions are approached from above rather than from below.

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Cornelia Drees, Kevin Chapman, Erin Prenger, Leslie Baxter, Rama Maganti, Harold Rekate, Andrew Shetter, Maggie Bobrowitz and John F. Kerrigan

Object

This study aimed at identifying outcomes with respect to seizures, morbidity, and mortality in adult patients undergoing resective or Gamma Knife surgery (GKS) to treat intractable epilepsy associated with hypothalamic hamartoma (HH).

Methods

Adult patients undergoing surgical treatment for HH-related epilepsy were prospectively monitored at a single center for complications and seizure outcome by using a proprietary database. Preintervention and postintervention data for patients 18 years of age and older, and with at least 1 year of follow-up, were analyzed, with specific attention to seizure control, complications, hormonal status, and death.

Results

Forty adult patients were found in the database (21 were women). The median HH volume was 0.54 cm3. In 70% of patients, it was located inside the third ventricle, attached unilaterally and vertically to the hypothalamus (Delalande Type II). Most patients (26) underwent an endoscopic resection, 10 patients had a transcallosal or other type of open (pterional or orbitozygomatic) resection, and 4 patients chose GKS.

Twenty-nine percent became seizure free in the long term, and overall a majority of patients (55%) reported at least > 90% seizure improvement. Only 3 patients were ultimately able to discontinue anticonvulsants, whereas most patients were taking an average of 2 antiepileptic drugs pre- and postoperatively. The only factor significantly correlated with seizure-free outcome was the absence of mental retardation. The HH volume, HH type, and amount of resection or disconnection were not correlated to seizure freedom. A total of 4 patients (10%) died, 2 immediately after surgery and 2 later. All of them had undergone a resection, as opposed to GKS, and still had seizures. Postoperatively, persistent neurological deficits were seen in 1 patient; 34% of patients had mild hormonal problems; and 59% experienced weight gain of at least 6.8 kg (average gain 12.7 kg).

Conclusions

Surgical or GKS procedures in adults with HH provided seizure freedom in one-third of patients. The only significant favorable prognostic factor was the absence of mental retardation. The overall mortality rate was high, at 10%. Other important morbidities were persistent hormonal disturbances and weight gain.

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Andrew S. Little, John F. Kerrigan, Cameron G. McDougall, Joseph M. Zabramski, Felipe C. Albuquerque, Peter Nakaji and Robert F. Spetzler

Object

Nonconvulsive status epilepticus (NCSE) is an underrecognized and poorly understood complication of aneurysmal subarachnoid hemorrhage (SAH). The authors evaluated the risk factors, electroencephalographic (EEG) characteristics, hospital course, and clinical outcomes associated with NCSE in a population with SAH treated at a single institution.

Methods

The hospitalization and outcome data were reviewed in 11 patients who had received a diagnosis of NCSE and SAH. The study included individuals from a cohort of 389 consecutive patients with SAH who were treated between March 2003 and June 2005, and who were analyzed retrospectively. The patients' medical history, neurological grade, events of hospitalization, EEG morphological patterns, and disposition were analyzed.

Advanced age, female sex, need for ventriculostomy, poor neurological grade (Hunt and Hess Grade III, IV, or V), thick cisternal blood clots, and structural lesions (intracerebral hemorrhage and stroke) were common in the population with NCSE. Patients with normal results on angiograms, good neurological grade (Hunt and Hess Grade I or II), and minimal SAH (Fisher Grade 1 or 2) were at lower risk. The most common ictal patterns were intermittent, and consisted of generalized periodic epileptiform discharges. Medical complications were also frequent, and the outcome of these patients was poor despite aggressive treatment regimens.

Conclusions

Nonconvulsive status epilepticus is a devastating complication of SAH with a high rate of associated morbidity. Based on these findings it appears that the patients at highest risk for NCSE can be identified, and this should provide a basis for further studies designed to determine the clinical significance of various EEG patterns and to develop preventative strategies.

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Adib A. Abla, Andrew G. Shetter, Steve W. Chang, Scott D. Wait, David G. Brachman, Yu-Tze Ng, Harold L. Rekate and John F. Kerrigan

Object

The authors present outcomes obtained in patients who underwent Gamma Knife surgery (GKS) at 1 institution as part of a multimodal treatment of refractory epilepsy caused by hypothalamic hamartomas (HHs).

Methods

Between 2003 and 2010, 19 patients with HH underwent GKS. Eight patients had follow-up for less than 1 year, and 1 patient was lost to follow-up. The 10 remaining patients (mean age 15.1 years, range 5.7–29.3 years) had a mean follow-up of 43 months (range 18–81 months) and are the focus of this report. Five patients had undergone a total of 6 prior surgeries: 1 transcallosal resection of the HH, 2 endoscopic transventricular resections of the HH, 2 temporal lobectomies, and 1 arachnoid cyst evacuation. In an institutional review board–approved study, postoperative complications and long-term outcome measures were monitored prospectively with the use of a proprietary database. Seven patients harbored Delalande Type II lesions; the remainder harbored Type III or IV lesions. Seizure frequency ranged from 1–2 monthly to as many as 100 gelastic seizures daily. The mean lesion volume was 695 mm3 (range 169–3000 mm3, median 265 mm3). The mean/median dose directed to the 50% isodose line was 18 Gy (range 16–20 Gy). The mean maximum point dose to the optic chiasm was 7.5 Gy (range 5–10 Gy). Three patients underwent additional resection 14.5, 21, and 32 months after GKS.

Results

Of the 10 patients included in this study, 6 are seizure free (2 after they underwent additional surgery), 1 has a 50%–90% reduction in seizure frequency, 2 have a 50% reduction in seizure frequency, and 1 has observed no change in seizure frequency. Overall quality of life, based on data obtained from follow-up telephone conversations and/or surveys, improved in 9 patients and was due to improvements in seizure control (9 patients), short-term memory loss (3 patients), and behavioral symptoms (5 patients); in 1 patient, quality of life remains minimally affected. Incidences of morbidity were all temporary and included poikilothermia (1 patient), increased depression (1 patient), weight gain/increased appetite (2 patients), and anxiety (1 patient) after GKS.

Conclusions

Of the approximately 150 patients at Barrow Neurological Institute who have undergone treatment for HH, the authors have reserved GKS for treatment of small HHs located distal from radiosensitive structures in patients with high cognitive function and a stable clinical picture, which allows time for the effects of radiosurgery to occur without further deterioration. The lack of significant morbidity and the clinical outcomes achieved in this study demonstrated a low risk of GKS for HH with results comparable to those of previous series.

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Adib A. Abla, Scott D. Wait, Jonathan A. Forbes, Sandipan Pati, Roger E. Johnsonbaugh, John F. Kerrigan and Yu-Tze Ng

Object

In this paper, the authors' goal was to describe the occurrence of alternating hypernatremia and hyponatremia in pediatric patients who underwent resection of hypothalamic hamartomas (HHs) for epilepsy. Hypernatremia in patients after pituitary or hypothalamic surgery can be caused by diabetes insipidus (DI), whereas hyponatremia can occur due to a syndrome of inappropriate antidiuretic hormone, cerebral salt wasting, or excessive administration of desmopressin (DDAVP). The triphasic response after surgery in the pituitary region can also explain variations in sodium parameters in such cases.

Methods

One hundred fifty-three patients with HH who underwent surgery were enrolled in a prospective study to monitor outcomes. Of these, 4 patients (2.6%) were noted to experience dramatic alterations in serum sodium values. The medical records of these patients were identified and evaluated.

Results

Patients' ages at surgery ranged from 1.2 to 6.0 years. All patients were girls. Two patients had Delalande Type IV lesions (of 16 total Type IV lesions surgically treated) and 2 had Type III lesions (of 39 total Type III lesions). All patients had a history of gelastic seizures refractory to medication. Seizure frequency ranged from 3 to 300 per day. After surgery, all patients experienced hypernatremia and hyponatremia. The largest fluctuation in serum sodium concentration during hospitalization in a single patient was 53 mEq/L (range 123–176 mEq/L). The mean absolute difference in maximum and minimum sodium values was 38.2 mEq/L.

All patients exhibited an initial period of immediate DI (independent of treatment) after surgery followed by a period of hyponatremia (independent of treatment), with a minimum value occurring between postoperative Days 5 and 8. All patients then returned to a hypernatremic state of DI, and 3 patients still require DDAVP for DI management. A second occurrence of hyponatremia lasting several days without DDAVP administration occurred in 2 patients during their hospitalization between periods of hypernatremia. One patient stabilized in the normal range of sodium values prior to discharge from rehabilitation without the need for further intervention. At last follow-up, 3 patients are seizure-free.

Conclusions

Severe instability of sodium homeostasis with hypernatremia and hyponatremia is seen in up to 2.6% of children undergoing open resection of HH. This risk appears to be related to HH type, with a higher risk for Types III (2 [5.1%] of 39) and IV (2 [12.5%] of 16) lesions. Here, the authors describe alternating episodes of hypernatremia and hyponatremia in the postoperative period following HH surgery. Management of this entity requires careful serial assessment of volume status and urine concentration and will often require alternating salt replacement therapy with DDAVP administration.

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Varina L. Boerwinkle, Lucia Mirea, William D. Gaillard, Bethany L. Sussman, Diana Larocque, Alexandra Bonnell, Jennifer S. Ronecker, Matthew M. Troester, John F. Kerrigan, Stephen T. Foldes, Brian Appavu, Randa Jarrar, Korwyn Williams, Angus A. Wilfong and P. David Adelson

OBJECTIVE

The authors’ goal was to prospectively quantify the impact of resting-state functional MRI (rs-fMRI) on pediatric epilepsy surgery planning.

METHODS

Fifty-one consecutive patients (3 months to 20 years old) with intractable epilepsy underwent rs-fMRI for presurgical evaluation. The team reviewed the following available diagnostic data: video-electroencephalography (n = 51), structural MRI (n = 51), FDG-PET (n = 42), magnetoencephalography (n = 5), and neuropsychological testing (n = 51) results to formulate an initial surgery plan blinded to the rs-fMRI findings. Subsequent to this discussion, the connectivity results were revealed and final recommendations were established. Changes between pre– and post–rs-fMRI treatment plans were determined, and changes in surgery recommendation were compared using McNemar’s test.

RESULTS

Resting-state fMRI was successfully performed in 50 (98%) of 51 cases and changed the seizure onset zone localization in 44 (88%) of 50 patients. The connectivity results prompted 6 additional studies, eliminated the ordering of 11 further diagnostic studies, and changed the intracranial monitoring plan in 10 cases. The connectivity results significantly altered surgery planning with the addition of 13 surgeries, but it did not eliminate planned surgeries (p = 0.003). Among the 38 epilepsy surgeries performed, the final surgical approach changed due to rs-fMRI findings in 22 cases (58%), including 8 (28%) of 29 in which extraoperative direct electrical stimulation mapping was averted.

CONCLUSIONS

This study demonstrates the impact of rs-fMRI connectivity results on the decision-making for pediatric epilepsy surgery by providing new information about the location of eloquent cortex and the seizure onset zone. Additionally, connectivity results may increase the proportion of patients considered eligible for surgery while optimizing the need for further testing.

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Oral Presentations

2010 AANS Annual Meeting Philadelphia, Pennsylvania May 1–5, 2010