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Yifei Duan, Jennifer Sweet, Charles Munyon and Jonathan Miller

OBJECT

Trigeminal neuralgia is often associated with nerve atrophy, in addition to vascular compression. The authors evaluated whether cross-sectional areas of different portions of the trigeminal nerve on preoperative imaging could be used to predict outcome after microvascular decompression (MVD).

METHODS

A total of 26 consecutive patients with unilateral Type 1a trigeminal neuralgia underwent high-resolution fast-field echo MRI of the cerebellopontine angle followed by MVD. Preoperative images were reconstructed and reviewed by 2 examiners blinded to the side of symptoms and clinical outcome. For each nerve, a computerized automatic segmentation algorithm was used to calculate the coronal cross-sectional area at the proximal nerve near the root entry zone and the distal nerve at the exit from the porus trigeminus. Findings were correlated with outcome at 12 months.

RESULTS

After MVD, 17 patients were pain free and not taking medications compared with 9 with residual pain. Across all cases, the coronal cross-sectional area of the symptomatic trigeminal nerve was significantly smaller than the asymptomatic side in the proximal part of the nerve, which was correlated with degree of compression at surgery. Atrophy of the distal trigeminal nerve was more pronounced in patients who had residual pain than in those with excellent outcome. Among the 7 patients who had greater than 20% loss of nerve volume in the distal nerve, only 2 were pain free and not taking medications at long-term follow-up.

CONCLUSIONS

Trigeminal neuralgia is associated with atrophy of the root entry zone of the affected nerve compared with the asymptomatic side, but volume loss in different segments of the nerve has very different prognostic implications. Proximal atrophy is associated with vascular compression and correlates with improved outcome following MVD. However, distal atrophy is associated with a significantly worse outcome after MVD.

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Suraj Suresh, Jennifer Sweet, Philip S. Fastenau, Hans Lüders, Patrick Landazuri and Jonathan Miller

OBJECT

Temporal lobe epilepsy (TLE) in the absence of MRI abnormalities and memory deficits is often presumed to have an extramesial or even extratemporal source. In this paper the authors report the results of a comprehensive stereoelectroencephalography (SEEG) analysis in patients with TLE with normal MRI images and memory scores.

METHODS

Eighteen patients with medically refractory epilepsy who also had unremarkable MR images and normal verbal and visual memory scores on neuropsychological testing were included in the study. All patients had seizure semiology and video electroencephalography (EEG) findings suggestive of TLE. A standardized SEEG investigation was performed for each patient with electrodes implanted into the mesial and lateral temporal lobe, temporal tip, posterior temporal neocortex, orbitomesiobasal frontal lobe, posterior cingulate gyrus, and insula. This information was used to plan subsequent surgical management.

RESULTS

Interictal SEEG abnormalities were observed in the mesial temporal structures in 17 patients (94%) and in the temporal tip in 6 (33%). Seizure onset was exclusively from mesial structures in 13 (72%), exclusively from lateral temporal cortex and/or temporal tip structures in 2 (11%), and independently from mesial and neocortical foci in 3 (17%). No seizure activity was observed arising from any extratemporal location. All patients underwent surgical intervention targeting the temporal lobe and tailored to the SEEG findings, and all experienced significant improvement in seizure frequency with a postoperative follow-up observation period of at least 1 year.

CONCLUSIONS

This study demonstrates 3 important findings: 1) normal memory does not preclude mesial temporal seizure onset; 2) onset of seizures exclusively from mesial temporal structures without early neocortical involvement is common, even in the absence of memory deficits; and 3) extratemporal seizure onset is rare when video EEG and semiology are consistent with focal TLE.

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Joshua J. Wind, P. Benjamin Kerr, Jennifer A. Sweet and Vivek R. Deshmukh

Pleomorphic xanthoastrocytomas are glial-based tumors that arise most frequently in young patients and usually follow a more benign and indolent clinical course than their other glial-based tumor counterparts. These tumors most frequently present with seizures, and only 3 previous case reports exist of hemorrhagic tumor as the clinical presentation. The authors present the first case of life-threatening intracerebral hemorrhage from pleomorphic xanthoastrocytoma in a child.

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Jennifer Sweet, Joshua Ammerman, Vivek Deshmukh and Joseph White

Cruciate paralysis is a clinical phenomenon thought to result from injury to decussating pyramidal tract fibers at the cervicomedullary junction, producing clinical findings of upper-extremity weakness out of proportion to the lower extremities. The authors present, to their knowledge, the first reported case of cruciate paralysis resulting from atlantooccipital dislocation.

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Christina Huang Wright, Dorian Kusyk, William S. Rosenberg and Jennifer A. Sweet

Lymphangiomatosis is a rare congenital disorder that results in multiorgan system lymphatic invasion. Symptoms due to axial skeletal involvement can range from chronic bone pain to severe deformity resulting in radiculopathy, myelopathy, and even paralysis. The authors present a case of lymphangiomatosis of the clivus, C-1, and C-2, resulting in chronic pain. The patient was successfully treated with percutaneous transoral clivoplasty and vertebroplasty, without disease progression or return of symptoms at 2 years.

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Asem Salma and Patrick T. Tracy

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Jennifer A. Sweet, Benjamin L. Walter, Kabilar Gunalan, Ashutosh Chaturvedi, Cameron C. McIntyre and Jonathan P. Miller

Object

Stimulation of white matter pathways near targeted structures may contribute to therapeutic effects of deep brain stimulation (DBS) for patients with Parkinson disease (PD). Two tracts linking the basal ganglia and cerebellum have been described in primates: the subthalamopontocerebellar tract (SPCT) and the dentatothalamic tract (DTT). The authors used fiber tractography to evaluate white matter tracts that connect the cerebellum to the region of the basal ganglia in patients with PD who were candidates for DBS.

Methods

Fourteen patients with advanced PD underwent 3-T MRI, including 30-directional diffusion-weighted imaging sequences. Diffusion tensor tractography was performed using 2 regions of interest: ipsilateral subthalamic and red nuclei, and contralateral cerebellar hemisphere. Nine patients underwent subthalamic DBS, and the course of each tract was observed relative to the location of the most effective stimulation contact and the volume of tissue activated.

Results

In all patients 2 distinct tracts were identified that corresponded closely to the described anatomical features of the SPCT and DTT, respectively. The mean overall distance from the active contact to the DTT was 2.18 ± 0.35 mm, and the mean proportional distance relative to the volume of tissue activated was 1.35 ± 0.48. There was a nonsignificant trend toward better postoperative tremor control in patients with electrodes closer to the DTT.

Conclusions

The SPCT and the DTT may be related to the expression of symptoms in PD, and this may have implications for DBS targeting. The use of tractography to identify the DTT might assist with DBS targeting in the future.

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Joshua J. Wind, Kamran D. Bakhtian, Jennifer A. Sweet, Gautam U. Mehta, Jayesh P. Thawani, Ashok R. Asthagiri, Edward H. Oldfield and Russell R. Lonser

Object

Brainstem hemangioblastomas are frequently encountered in patients with von Hippel-Lindau (VHL) disease. These tumors can cause significant morbidity, and their optimal management has not been defined. To better define the outcome and management of these tumors, the authors analyzed the long-term results in patients who underwent resection of brainstem hemangioblastomas.

Methods

Consecutive patients with VHL disease who underwent resection of brainstem hemangioblastomas with a follow-up of 12 months or more were included in this study. Serial functional assessments, radiographic examinations, and operative records were analyzed.

Results

Forty-four patients (17 male and 27 female) underwent 51 operations for resection of 71 brainstem hemangioblastomas. The most common presenting symptoms were headache, swallowing difficulties, singultus, gait difficulties, and sensory abnormalities. The mean follow-up was 5.9 ± 5.0 years (range 1.0–20.8 years). Immediately after 34 operations (66.7%), the patients remained at their preoperative functional status; they improved after 8 operations (15.7%) and worsened after 9 operations (17.6%) as measured by the McCormick scale. Eight (88.9%) of the 9 patients who were worse immediately after resection returned to their preoperative status within 6 months. Two patients experienced functional decline during long-term follow-up (beginning at 2.5 and 5 years postoperatively) caused by extensive VHL disease–associated CNS disease.

Conclusions

Generally, resection of symptomatic brainstem hemangioblastomas is a safe and effective management strategy in patients with VHL disease. Most patients maintain their preoperative functional status, although long-term decline in functional status may occur due to VHL disease–associated progression.

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Jun T. Park, Sunil V. Manjila, Rachel B. Tangen, Mark L. Cohen, Asim M. Shahid, Jennifer A. Sweet, Ingrid E. Tuxhorn and Jonathan P. Miller

Catastrophic epilepsy in infants, often due to extensive cortical dysplasia, has devastating consequences with respect to brain development. Conventional lobar, multilobar, or hemispheric resection in these infants is challenging, carrying an increased operative risk compared with that in older children. Removing a larger tissue volume versus removing or disconnecting the epileptogenic region does not always guarantee better seizure outcome. The authors describe 2 infants with catastrophic epilepsy who benefited from individually tailored disconnections based on a hypothesized epileptogenic zone following intensive presurgical evaluation.

Two infants with catastrophic epilepsy and epileptic spasms underwent leukotomies between 3 and 12 months of age. They were followed up postoperatively for 19–36 months. Both patients had 90%–100% seizure reduction and a significantly improved neurodevelopmental outcome without postoperative complication. Cortical malformation was seen in both patients. Modifications of established surgical disconnection techniques, tailored to each patient's specific epileptogenic zone, optimized seizure and neurodevelopmental outcomes while minimizing the risks associated with more extensive resections.