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Kelsey Cobourn, Islam Fayed, Robert F. Keating and Chima O. Oluigbo

OBJECTIVE

Stereoelectroencephalography (sEEG) and MR-guided laser interstitial thermal therapy (MRgLITT) have both emerged as minimally invasive alternatives to open surgery for the localization and treatment of medically refractory lesional epilepsy. Although some data are available about the use of these procedures individually, reports are almost nonexistent on their use in conjunction. The authors’ aim was to report early outcomes regarding efficacy and safety of sEEG followed by MRgLITT for localization and ablation of seizure foci in the pediatric population with medically refractory lesional epilepsy.

METHODS

A single-center retrospective review of pediatric patients who underwent sEEG followed by MRgLITT procedures was performed. Demographic, intraoperative, and outcome data were compiled and analyzed.

RESULTS

Four pediatric patients with 9 total lesions underwent sEEG followed by MRgLITT procedures between January and September 2017. The mean age at surgery was 10.75 (range 2–21) years. Two patients had tuberous sclerosis and 2 had focal cortical dysplasia. Methods of stereotaxy consisted of BrainLab VarioGuide and ROSA robotic guidance, with successful localization of seizure foci in all cases. The sEEG procedure length averaged 153 (range 67–235) minutes, with a mean of 6 (range 4–8) electrodes and 56 (range 18–84) contacts per patient. The MRgLITT procedure length averaged 223 (range 179–252) minutes. The mean duration of monitoring was 6 (range 4–8) days, and the mean total hospital stay was 8 (range 5–11) days. Over a mean follow-up duration of 9.3 (range 5.1–16) months, 3 patients were seizure free (Engel class I, 75%), and 1 patient saw significant improvement in seizure frequency (Engel class II, 25%). There were no complications.

CONCLUSIONS

These early data demonstrate that sEEG followed by MRgLITT can be used safely and effectively to localize and ablate epileptogenic foci in a minimally invasive paradigm for treatment of medically refractory lesional epilepsy in pediatric populations. Continued collection of data with extended follow-up is needed.

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Kee Kiat Yeo, Elena Puscasiu, Robert F. Keating and Brian R. Rood

Cellular hemangioma is a subtype of hemangioma that is associated with cellular immaturity and the potential for recurrence. Intracranial location of these lesions is extremely rare, and definitive treatment often requires radical neurosurgical resection. The authors report a case of a 12-year-old boy with a subtemporal cellular hemangioma. He underwent gross-total resection of the tumor, but within 1.5 months the tumor recurred, necessitating a second resection. Because of its proximity to vascular structures, only subtotal resection was possible. Repeat MRI 1 month after the second surgery showed significant tumor recurrence. Given the tumor's demonstrated capacity for recurrence and its proximity to the vein of Labbé and sigmoid sinus, further resection was not indicated. In an effort to limit radiation therapy for this young patient, treatment with bevacizumab and temozolomide was chosen and achieved a complete response that has proven durable for 36 months after cessation of therapy. This is the first report of the successful use of chemotherapy to treat an intracranial hemangioma, a rare condition with limited therapeutic options.

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Editorial

Posterior fossa syndrome

Mark M. Souweidane

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Mitchel Seruya, Albert K. Oh, Michael J. Boyajian, Jeffrey C. Posnick and Robert F. Keating

Object

Patients with delayed presentation of isolated sagittal synostosis (ISS) pose unique surgical challenges. Intracranial hypertension can be missed in the absence of overt findings. Here, an algorithm is presented for the elucidation of intracranial hypertension and approaches to surgical treatment during calvarial reconstruction.

Methods

Patients with delayed presentation (age > 15 months) of ISS between 1997 and 2009 were identified. Symptoms, signs, and radiological evidence of intracranial hypertension were noted. Intraoperative management included calvarial reconstruction in conjunction with ventriculostomy for intracranial pressure (ICP) monitoring and CSF drainage in the setting of suspected intracranial hypertension.

Results

Seventeen patients underwent calvarial reconstruction for delayed presentation of ISS. The mean surgical age was 40.5 months (16.2–82.9 months), and the average follow-up was 34.2 months (0.6–92.2 months). Eleven patients with subtle findings of intracranial hypertension underwent ICP monitoring during calvarial reconstruction. The mean opening ICP was 23.5 cm H2O (16.5–29.5 cm H2O), and the mean closing ICP was 7.0 cm H2O (3.5–17.0 cm H2O). Nine (81.8%) of 11 monitored patients demonstrated intracranial hypertension (ICP ≥ 20 cm H2O); the other 2 had borderline increased ICP. Perioperative morbidity was 5.9%, with 1 patient medically treated for transient, new-onset intracranial hypertension. The postoperative Whitaker category was I in 94.1% of patients, II in 5.9%, and III/IV in 0%. There were no reoperations or deaths.

Conclusions

In this consecutive series, 11 of 17 patients with delayed presentation of ISS underwent ICP monitoring during calvarial reconstruction as a result of subtle clinical findings of intracranial hypertension. Nine (81.8%) of 11 monitored patients demonstrated intracranial hypertension. Calvarial reshaping along with ICP monitoring and CSF drainage facilitated reconstruction and resulted in good outcomes and the resolution of intracranial hypertension.

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Brian R. Curtis, Rory J. Petteys, Christopher T. Rossi, Robert F. Keating and Suresh N. Magge

Aneurysmal bone cyst (ABC) is an uncommon benign, tumorlike lesion of bone that is usually located in the long bones and spine. On rare occasions, ABCs are found in the bones of the cranial vault and skull base, occasionally causing mass effect and cranial nerve findings. In this report the authors detail the case of a patient who presented with incidentally discovered hydrocephalus due to a large ABC of the occipital bone that produced mass effect and obstruction of CSF. The diagnosis, imaging findings, and surgical management of this interesting and rare case are discussed.

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Benjamin C. Wood, Albert K. Oh, Robert F. Keating, Michael J. Boyajian, John S. Myseros, Suresh N. Magge and Gary F. Rogers

OBJECT

Progressive postnatal pansynostosis (PPP) is a rare form of craniosynostosis that is characterized by a normal head shape, insidious decrease in percentile head circumference, and high rates of elevated intracranial pressure (ICP). This investigation describes the clinical, radiographic, and genetic features of this entity.

METHODS

The authors’ craniofacial database for the period 1997–2013 was retrospectively culled to identify patients who had a normal or near-normal head shape and CT-confirmed multiple-suture synostosis. Patients with kleeblatt-schädel or previous craniofacial surgery were excluded. All demographic information was collected and analyzed.

RESULTS

Seventeen patients fit the inclusion criteria. Nine patients had a syndromic diagnosis: Crouzon syndrome (n = 4), Pfeiffer syndrome (n = 2), Saethre-Chotzen syndrome (n = 1), Apert syndrome (n = 1), and achondroplasia (n = 1). With the exception of 3 patients with mild turricephaly, all patients had a relatively normal head shape. Patients were diagnosed at an average age of 62.9 months. Nearly all patients had some combination of clinical, radiographic, or ophthalmological evidence of increased ICP.

CONCLUSIONS

PPP is insidious; diagnosis is typically delayed because the clinical signs are subtle and appear gradually. All normocephalic infants or children with a known or suspected craniosynostotic disorder should be carefully monitored; any decrease in percentile head circumference or signs/symptoms of increased ICP should prompt CT evaluation.

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Matthew F. Sacino, Cheng-Ying Ho, Jonathan Murnick, Robert F. Keating, William D. Gaillard and Chima O. Oluigbo

OBJECTIVE

Previous studies have demonstrated that an important factor in seizure freedom following surgery for lesional epilepsy in the peri-eloquent cortex is completeness of resection. However, aggressive resection of epileptic tissue localized to this region must be balanced with the competing objective of retaining postoperative neurological functioning. The objective of this study was to investigate the role of intraoperative MRI (iMRI) as a complement to existing epilepsy protocol techniques and to compare rates of seizure freedom and neurological deficit in pediatric patients undergoing resection of perieloquent lesions.

METHODS

The authors retrospectively reviewed the medical records of pediatric patients who underwent resection of focal cortical dysplasia (FCD) or heterotopia localized to eloquent cortex regions at the Children's National Health System between March 2005 and August 2015. Patients were grouped into two categories depending on whether they underwent conventional resection (n = 18) or iMRI-assisted resection (n = 11). Patient records were reviewed for factors including demographics, length of hospitalization, postoperative seizure freedom, postoperative neurological deficit, and need for reoperation. Postsurgical seizure outcome was assessed at the last postoperative follow-up evaluation using the Engel Epilepsy Surgery Outcome Scale.

RESULTS

At the time of the last postoperative follow-up examination, 9 (82%) of the 11 patients in the iMRI resection group were seizure free (Engel Class I), compared with 7 (39%) of the 18 patients in the control resection group (p = 0.05). Ten (91%) of the 11 patients in the iMRI cohort achieved gross-total resection (GTR), compared with 8 (44%) of 18 patients in the conventional resection cohort (p = 0.02). One patient in the iMRI-assisted resection group underwent successful reoperation at a later date for residual dysplasia, compared with 7 patients in the conventional resection cohort (with 2/7 achieving complete resection). Four (36%) of the patients in the iMRI cohort developed postoperative neurological deficits, compared with 15 patients (83%) in the conventional resection cohort (p = 0.02).

CONCLUSIONS

These results suggest that in comparison with a conventional surgical protocol and technique for resection of epileptic lesions in peri-eloquent cortex, the incorporation of iMRI led to elevated rates of GTR and postoperative seizure freedom. Furthermore, this study suggests that iMRI-assisted surgeries are associated with a reduction in neurological deficits due to intraoperative damage of eloquent cortex.

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Benjamin C. Wood, Edward S. Ahn, Joanna Y. Wang, Albert K. Oh, Robert F. Keating, Gary F. Rogers and Suresh N. Magge

OBJECTIVE

Endoscopic strip craniectomy (ESC) with postoperative helmet orthosis is a well-established treatment option for sagittal craniosynostosis. There are many technical variations to the surgery ranging from simple strip craniectomy to methods that employ multiple cranial osteotomies. The purpose of this study was to determine whether the addition of lateral barrel-stave osteotomies during ESC improved morphological outcomes.

METHODS

An IRB-approved retrospective review was conducted on a consecutive series of cases involving ESC for sagittal craniosynostosis at 2 different institutions from March 2008 to August 2014. The patients in Group A underwent ESC and those in Group B had ESC with lateral barrel-stave osteotomies. Demographic and perioperative data were recorded; postoperative morphological outcomes were analyzed using 3D laser scan data acquired from a single orthotic manufacturer who managed patients from both institutions.

RESULTS

A total of 73 patients were included (34 in Group A and 39 in Group B). Compared with Group B patients, Group A patients had a shorter mean anesthetic time (161.7 vs 195 minutes; p < 0.01) and operative time (71.6 vs 111 minutes; p < 0.01). The mean hospital stay was similar for the 2 groups (1.2 days for Group A vs 1.4 days for Group B; p = 0.1). Adequate postoperative data on morphological outcomes were reported by the orthotic manufacturer for 65 patients (29 in Group A and 36 in Group B). The 2 groups had similar improvement in the cephalic index (CI): Group A, mean change 10.5% (mean preoperative CI 72.6, final 80.4) at a mean follow-up of 13.2 months; Group B, mean change 12.2% (mean preoperative CI 71.0, final 79.6) at a mean follow-up of 19.4 months. The difference was not statistically significant (p = 0.15).

CONCLUSIONS

Both ESC alone and ESC with barrel staving produced excellent outcomes. However, the addition of barrel staves did not improve the results and, therefore, may not be warranted in the endoscopic treatment of sagittal craniosynostosis.

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Alan Siu, Gary F. Rogers, John S. Myseros, Siri S. Khalsa, Robert F. Keating and Suresh N. Magge

There is no known correlation between Down syndrome and craniosynostosis. The authors report 2 infants with trisomy 21 and right unilateral coronal craniosynostosis. Both patients were clinically asymptomatic but displayed characteristic craniofacial features associated with each disorder. One patient underwent a bilateral fronto-orbital advancement and the other underwent an endoscopically assisted strip craniectomy with postoperative helmet therapy. Both patients demonstrated good cosmesis at follow-up.

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Siri Sahib S. Khalsa, Alan Siu, Tiffani A. DeFreitas, Justin M. Cappuzzo, John S. Myseros, Suresh N. Magge, Chima O. Oluigbo and Robert F. Keating

OBJECTIVE

Previous studies have indicated an association of Chiari malformation Type I (CM-I) and a small posterior fossa. Most of these studies have been limited by 2D quantitative methods, and more recent studies utilizing 3D methodologies are time-intensive with manual segmentation. The authors sought to develop a more automated tool to calculate the 3D posterior fossa volume, and correlate its changes after decompression with surgical outcomes.

METHODS

A semiautomated segmentation program was developed, and used to compare the pre- and postoperative volumes of the posterior cranial fossa (PCF) and the CSF spaces (cisterna magna, prepontine cistern, and fourth ventricle) in a cohort of pediatric patients with CM-I. Volume changes were correlated with postoperative symptomatic improvements in headache, syrinx, tonsillar descent, cervicomedullary kinking, and overall surgical success.

RESULTS

Forty-two pediatric patients were included in this study. The mean percentage increase in PCF volume was significantly greater in patients who showed clinical improvement versus no improvement in headache (5.89% vs 1.54%, p < 0.05) and tonsillar descent (6.52% vs 2.57%, p < 0.05). Overall clinical success was associated with a larger postoperative PCF volume increase (p < 0.05). These clinical improvements were also significantly associated with a larger increase in the volume of the cisterna magna (p < 0.05). The increase in the caudal portion of the posterior fossa volume was also larger in patients who showed improvement in syrinx (6.63% vs 2.58%, p < 0.05) and cervicomedullary kinking (9.24% vs 3.79%, p < 0.05).

CONCLUSIONS

A greater increase in the postoperative PCF volume, and specifically an increase in the cisterna magna volume, was associated with a greater likelihood of clinical improvements in headache and tonsillar descent in patients with CM-I. Larger increases in the caudal portion of the posterior fossa volume were also associated with a greater likelihood of improvement in syrinx and cervicomedullary kinking.