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  • Author or Editor: Scott D. Wait x
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Douglas R. Taylor, Scott D. Wait, James W. Wheless and Frederick A. Boop

Neurocutaneous melanosis (NCM) is a rare, congenital neuroectodermal dysplasia generally resulting in early death from neurological decline due to leptomeningeal involvement. Nonmeningeal CNS epileptogenic lesions presenting in later childhood in the absence of leptomeningeal disease are rare. This report summarizes a rare presentation and curative epilepsy surgery.

The authors discuss the case of a 14-year-old girl with NCM who originally presented with intractable partial-onset seizures. The MRI, PET, and SPECT studies subsequently revealed a focal epileptogenic source in the right temporal lobe. Results of video-electroencephalography monitoring concurred with the imaging findings, and a right temporal lobectomy was performed including the medial structures. Following surgery, histopathological features of the lesion included multiple scattered mononuclear cells with brown pigmentation in the amygdala specimen. The patient remains seizure free 2 years postresection, and no longer needs medication for seizure management.

This patient presented with an atypical CNS manifestation of NCM that is curable by epilepsy surgery. Her intractable epilepsy developed secondary to amygdalar neuromelanosis, which had no associated leptomeningeal melanosis, an uncommon occurrence. As evidenced by the lack of seizure activity following resection, the patient's quality of life greatly improved after neurological surgery.

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Weier Li, Scott D. Wait, Robert J. Ogg, Matt A. Scoggins, Ping Zou, James Wheless and Frederick A. Boop

Object

Advances in brain imaging have allowed for more sophisticated mapping of crucial neural structures. Functional MRI (fMRI) measures local changes in blood oxygenation associated with changes in neural activity and is useful in mapping cortical activation. Applications of this imaging modality have generally been restricted to cooperative patients; however, fMRI has proven successful in localizing the motor cortex for neurosurgical planning in uncooperative children under sedation. The authors demonstrate that the use of fMRI to localize the visual cortex in sedated children can be safely and effectively performed, allowing for more accurate presurgical planning to spare visual structures.

Methods

Between 2007 and 2009, 11 children (age range 1–11 years) underwent fMRI for neurosurgical planning while under sedation. Blood oxygen level–dependent fMRI was performed to detect visual cortex activation during stimulation through closed eyelids. Visual stimulation was presented in block design with periods of flashing light alternated with darkness.

Results

Functional MRI was successful in identifying visual cortex in each of the 11 children tested. There were no complications with propofol sedation or the fMRI. All children suffered from epilepsy, 5 had brain tumors, and 1 had tuberous sclerosis. After fMRI was performed, 6 patients underwent surgery. Frameless stereotactic guidance was synchronized with fMRI data to design an approach to spare visual structures during resection. There were no cases where a false negative led to unexpected visual field deficits or other side effects of surgery. In 2 cases, the fMRI results demonstrated that the tracts were already disrupted: in one case from a prior tumor operation and in another from dysplasia.

Conclusions

Functional MRI for evaluation of visual pathways can be safely and reproducibly performed in young or uncooperative children under light sedation. Identification of primary visual cortex aids in presurgical planning to avoid vision loss in appropriately selected patients.