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ABSTRACT: Craniopharyngioma is histologically benign and associated with high survival rates but poor quality of life. The SEER Program is among the most cited data sources regarding malignancies in the United States. SEER began collecting data regarding craniopharyngiomain 2004. SEER-STAT v7.0.5 was utilized to identify patients (January 1, 2004-December 31, 2008) with ICD-O-3 codes for craniopharyngioma. Age was categorized into 3 groups: ≤19, 20-34, and ≥35 years, as was surgical intervention: none, subtotal resection, and gross total/radical resection. Demographic, initial treatment, and follow-up data were collected. 635/662 (95.9 %) patients had complete data. Incidence per million patient-years by age group was 1.9, 1.1, and 1.9, respectively (p < 0.0001). There was bimodal incidence, with peaks at 5-9 and 60-74 years. Surgery occurred in 528 patients (83.1 %), without association between age group and extent of surgery (p = 0.14). Radiation was delivered in 139 (21.9 %) cases, with no association between treatment and extent of surgery (p = 0.73) or age group (p = 0.14). Median follow-up was 23 months. Overall and Cause-specific Survival were 87.9 and 94.5 %, respectively. Neither was associated with extent of surgery but both were positively associated with radiation (p = 0.0003 and 0.0007, respectively). There was no difference in OS or CSS when comparing STR ± RT versus GTR alone (p = 0.38 and 0.56, respectively). SEER provides reliable demographic and survival data regarding craniopharyngioma. SEER's focus on mortality statistics limits utility for outcomes studies in tumors with high survival rates, such as craniopharyngioma. Initial treatment data from SEER varies somewhat from current literature, meriting further investigation.
Journal of Neuro-Oncology 08/2012; 110(2):271-8. · 3.21 Impact Factor
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ABSTRACT: The diagnosis of diffuse pontine tumors has largely been made on the basis of MRI since the early 1990 s. In cases of tumors considered "typical," as a rule, no biopsy specimen has been obtained, and the tumors have been considered diffuse intrinsic pontine gliomas (DIPGs). There have been sporadic reports that primitive neuroectodermal tumors (PNETs) of the pons may not be distinguishable from the DIPGs by radiological imaging. This study presents 2 cases of diffuse pontine PNETs with molecular evidence that these are indeed PNETs, distinct from DIPGs, thus supporting biopsy of diffuse pontine tumors as a standard of care.
Biopsy specimens were obtained from 7 diffuse pontine tumors and snap frozen. Two of these 7 tumors were identified on the basis of pathological examination as PNETs. All 7 of the diffuse pontine tumors were analyzed for gene expression using the Affymetrix HG-U133 Plus 2.0 GeneChip microarray. Gene expression was compared with that of supratentorial PNETs, medulloblastomas, and low- and high-grade gliomas outside the brainstem.
Unsupervised hierarchical clustering analysis of gene expression demonstrated that pontine PNETs are most closely related to PNETs of the supratentorial region and not with gliomas. They do not cluster with the 5 DIPGs in the study. Thirty-eight genes, including GATA3, are uniquely differentially expressed in pontine PNETs compared with other types of pediatric brain tumors, including DIPGs and other PNETs at a false discovery rate statistical significance of less than 0.05.
The cluster and individual gene expression analyses indicate that pontine PNETs are intrinsically different from DIPGs. The 2 pontine PNET cases cluster with supratentorial PNETs, rather than with DIPGs, suggesting that these tumors should be treated with a PNET regimen, not with DIPG therapy. Since diagnosis by imaging is not reliable and the biology of the tumors is disparate, a biopsy should be performed to enable accurate diagnosis and direct potentially more effective treatments.
Journal of Neurosurgery Pediatrics 06/2012; 10(2):81-8. · 1.53 Impact Factor
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ABSTRACT: Atypical teratoid/rhabdoid tumor (AT/RT) is an uncommon, aggressive, embryonal pediatric brain tumor that almost always develops de novo and does not arise within, or evolve from, other brain tumor types. Although rhabdoid morphology can be seen in other tumor types, these are phenotypic mimics and, with only rare exceptions, do not manifest the INI-1 deletion at the 22q11.2 locus or the INI-1 nuclear protein loss that characterizes AT/RT. A few reports of AT/RT evolving from a low-grade ganglioglioma (GG) or pleomorphic xanthoastrocytoma have appeared. We present the case of a 6-year-old boy with a large right parietal mass whose tumor at initial presentation manifested 2 distinct components: GG with neoplastic neurons, low MIB-1 rate, and retention of INI-1 nuclear immunostaining (immunohistochemical) and, second, AT/RT with rhabdoid cells, polyphenotypic immunohistochemical expression, high MIB-1 rate, and loss of INI-1 nuclear expression. The 2 areas were separately assessed by fluorescence in situ hybridization for monosomy 22; monosomy 22 was identified in the AT/RT component but not in the GG areas. BRAF V600E mutation, a genetic abnormality seen in a significant percentage of pleomorphic xanthoastrocytomas and GGs, was assessed by polymerase chain reaction and identified in the tumor. Dual abnormalities of INI-1 loss and V600E BRAF mutation were identified in a cell culture line established from cerebrospinal fluid metastatic tumor cells. This cell line exhibited extremely rapid growth rate and rhabdoid morphology. Results suggest a postclonal modification in a subset of GG cells, with acquisition of INI-1 loss, confirming by biological methods what was previously suspected in rare reports of AT/RT evolving from other tumor types.
The American journal of surgical pathology 11/2011; 35(12):1894-901. · 4.06 Impact Factor
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ABSTRACT: Occipitoatlantoaxial rotatory fixation (OAARF) is a rare condition involving fixed rotational subluxation of the atlas in relation to both the occiput and axis. Atlantoaxial rotatory fixation (AARF) appears to precede OAARF in most cases, as untreated AARF may cause compensatory counter-rotation and occipitoaxial fixation at an apparently neutral head position. We report a case of OAARF in an 8-year-old girl with juvenile idiopathic arthritis. Cervical imaging demonstrated slight rightward rotation of the occiput at 7.63° in relation to C-2 and significant rightward rotation of C-1 at 65.90° in relation to the occiput and at 73.53° in relation to C-2. An attempt at closed reduction with halo traction was unsuccessful. Definitive treatment included open reduction, C-1 laminectomy, and occipitocervical internal fixation and fusion.
Journal of Neurosurgery Pediatrics 08/2011; 8(2):198-204. · 1.53 Impact Factor
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ABSTRACT: The current management paradigm for children with newly diagnosed diffuse intrinsic pontine glioma (DIPG) is to establish a diagnosis and begin therapy based on MR imaging findings correlated with an appropriate clinical presentation, and without a tissue diagnosis. This strategy assumes that pediatric neurosurgeons and neurooncologists uniformly interpret MR imaging findings in this population. This study sought to examine the consistency of North American pediatric neurosurgeons in assessing MR images in this patient population, and in their surgical plans based on the interpretation of those images.
The authors created an online survey and invited all members of the American Society of Pediatric Neurosurgeons and the American Association of Neurological Surgeons/Congress of Neurological Surgeons Joint Section for Pediatric Neurosurgery to participate. The survey consisted of 58 questions, and 48 pertained to representative MR images from 16 children who presented to The Children's Hospital of Colorado with diffuse pontine tumors. Based on the imaging presented and a standard clinical scenario, the respondent was asked if she or he believed a lesion to be "typical" or "atypical," whether she or he would biopsy the lesion, and what surgical approach would be chosen. The remaining 10 questions pertained to respondent demographics and his or her practice regarding tissue preservation and interest in participating in a multicenter trial that included tumor biopsy in selected cases. Rates at which each lesion was considered to be typical or atypical and rates of recommended biopsy were calculated.
Surveys were received by 269 individuals. Eighty-six responses were received (32.0%). No tumor was judged to be either typical or atypical by all respondents. Individual surgeons varied widely regarding how many of the tumors were judged as typical or warranted a biopsy. The percentage of respondents who disagreed with the majority opinion regarding whether a tumor qualified as typical ranged from 2.3% to 48.8%, with a median of 28.6%. More than 75% agreement regarding whether a tumor was typical or atypical was found in 7 (43.8%) of 16 cases. The κ statistic regarding typicality was 0.297 ± 0.0004 (mean ± SEM), implying only fair agreement. For every tumor, at least 1 respondent who believed the lesion to be typical in appearance would nevertheless biopsy the lesion. Of those respondents who considered a lesion to be typical, a median of 5.1% (range 1.2%-66.7%) would choose to biopsy this lesion. Of those respondents who considered a lesion atypical, a median of 18.3% (range 3.7%-100%) would choose not to biopsy the lesion. Of 85 responses to the question, "Would you be willing to biopsy a typical diffuse pontine glioma as a part of a multicenter trial?," 59 (69.4%) of 85 respondents answered that they would.
Although making a diagnosis based on radiographic evidence alone represents a well-established management paradigm for children with suspected DIPG, this study demonstrates considerable inconsistency on the part of pediatric neurosurgeons in the application of this strategy to individual patients. As such, the practice of diagnosing DIPG based on imaging characteristics and clinical history alone does not reach the appropriate threshold to be considered a standard of care.
Journal of Neurosurgery Pediatrics 07/2011; 8(1):97-102. · 1.53 Impact Factor
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ABSTRACT: Lymphatic malformations that involve the nervous system are uncommon. The authors review their experience with involvement of the brachial plexus and its branches by cystic hygromas. A retrospective review of the authors' experience with pathology of the pediatric brachial plexus revealed 4 cases involving patients with compression of this structure and its branches due to cystic hygroma. Although such cases are apparently rare, the neurosurgeon should consider malformations of the lymphatic system in the differential diagnosis of masses involving the brachial plexus and its branches.
Journal of Neurosurgery Pediatrics 03/2011; 7(3):282-5. · 1.53 Impact Factor
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ABSTRACT: The traditional reasons for surgical intervention in children with single-suture craniosynostosis (SSC) are cosmetic improvement and the avoidance/treatment of intracranial hypertension, which has been thought to contribute to neurocognitive deficits. Despite considerable work on the topic, the exact prevalence of intracranial hypertension in the population of patients with SSC is unknown, although it appears to be present in only a minority. Additionally, recent neuropsychological and anatomical literature suggests that the subtle neurocognitive deficits identified in children with a history of SSC may not result from external compression. They may instead reflect an underlying developmental condition that includes disordered primary CNS development and early suture fusion. This implies that current surgical techniques are unlikely to prevent neurocognitive deficits in patients with SSC. As such, the most common indication for surgical treatment in SSC is cosmetic, and most patients benefit from considerable subjective cosmetic normalization following surgery. Pediatric craniofacial surgeons have not, however, agreed upon objective means to assess postoperative cranial morphological improvement. We should therefore endeavor to agree upon objective craniometric tools for the assessment of operative outcomes, allowing us to accurately compare the various surgical techniques that are currently available.
Journal of Neurosurgery Pediatrics 08/2010; 6(2):193-7. · 1.53 Impact Factor
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ABSTRACT: In rare cases, children with a Chiari malformation Type I (CM-I) suffer from concomitant, irreducible, ventral brainstem compression that may result in cranial neuropathies or brainstem dysfunction. In these circumstances, a 360 degrees decompression supplemented by posterior stabilization and fusion is required. In this report, the authors present the first experience with using an endoscopic transnasal corridor to accomplish ventral decompression in children with CM-I that is complicated by ventral brainstem compression.
Two children presented with a combination of occipital headaches, swallowing dysfunction, myelopathy, and/or progressive scoliosis. Imaging studies demonstrated CM-I with severely retroflexed odontoid processes and ventral brainstem compression. Both patients underwent an endoscopic transnasal approach for ventral decompression, followed by posterior decompression, expansive duraplasty, and occipital-cervical fusion.
In both patients the endoscopic transnasal approach provided excellent ventral access to decompress the brainstem. When compared with the transoral approach, endoscopic transnasal access presents 4 potential advantages: 1) excellent prevertebral exposure in patients with small oral cavities; 2) a surgical corridor located above the hard palate to decompress rostral pathological entities more easily; 3) avoidance of the oral trauma and edema that follows oral retractor placement; and 4) avoidance of splitting the soft or hard palate in patients with oral-palatal dysfunction from ventral brainstem compression.
The endoscopic transnasal approach is atraumatic to the oral cavity, and offers a more superior region of exposure when compared with the standard transoral approach. Depending on their comfort level with endoscopic surgical techniques, pediatric neurosurgeons should consider this approach in children with pathological entities requiring ventral brainstem decompression.
Journal of Neurosurgery Pediatrics 06/2010; 5(6):549-53. · 1.53 Impact Factor
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Todd C Hankinson,
Anthony M Avellino,
David Harter,
Andrew Jea,
Sean Lew,
David Pincus,
Mark R Proctor,
Luis Rodriguez,
David Sacco,
Theodore Spinks,
Douglas L Brockmeyer,
Richard C E Anderson
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ABSTRACT: The object of this study was to assess a multiinstitutional experience with pediatric occipitocervical constructs to determine whether a difference exists between the fusion and complication rates of constructs with or without direct C-1 instrumentation.
Seventy-seven cases of occiput-C2 instrumentation and fusion, performed at 9 children's hospitals, were retrospectively analyzed. Entry criteria included atlantooccipital instability with or without atlantoaxial instability. Any case involving subaxial instability was excluded. Constructs were divided into 3 groups based on the characteristics of the anchoring spinal instrumentation: Group 1, C-2 instrumentation; Group 2, C-1 and C-2 instrumentation without transarticular screw (TAS) placement; and Group 3, any TAS placement. Groups were compared based on rates of fusion and perioperative complications.
Group 1 consisted of 16 patients (20.8%) and had a 100% rate of radiographically demonstrated fusion. Group 2 included 22 patients (28.6%), and a 100% fusion rate was achieved, although 2 cases were lost to follow-up before documented fusion. Group 3 included 39 patients (50.6%) and demonstrated a 100% radiographic fusion rate. Complication rates were 12.5, 13.7, and 5.1%, respectively. There were 3 vertebral artery injuries, 1 (4.5%) in Group 2 and 2 (5.1%) in Group 3.
High fusion rates and low complication rates were achieved with each configuration examined. There was no difference in fusion rates between the group without (Group 1) and those with (Groups 2 and 3) C-1 instrumentation. These findings indicated that in the pediatric population, excellent occipitocervical fusion rates can be accomplished without directly instrumenting C-1.
Journal of Neurosurgery Pediatrics 04/2010; 5(4):380-4. · 1.53 Impact Factor
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ABSTRACT: Children with Down syndrome may have occipitocervical and atlantoaxial instability. To prevent neurologic injury during athletic competitions, such as the Special Olympics, radiographic cervical spine screening was established in 1983 as a prerequisite for participation in some events. This review discusses the biomechanics underlying upper cervical instability in children with Down syndrome, the evolution of cervical spine screening protocols, and current opinion regarding management for children with Down syndrome and upper cervical instability.
Neurosurgery 03/2010; 66(3 Suppl):32-8. · 2.79 Impact Factor
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ABSTRACT: The capacity for local infection to prolong survival in patients with cancer is a widely accepted but unsubstantiated principle. The neurosurgical literature contains anecdotal reports of patients with malignant gliomas who experienced prolonged remission of their tumors after a bacterial infection. This association has not been explored in a larger series of patients with malignant glioma with postoperative infections.
A single-center operative experience accumulated over 10 years was examined to evaluate whether postoperative infections conferred a survival advantage in patients with glioblastoma multiforme. A total of 382 patients were examined, and 18 bacterial infections were identified. The vast majority (17 cases, 94%) of these were gram-positive infections. Cases were compared with age-matched controls. Survival differences were evaluated using Kaplan-Meier curves, and other differences were tested using the Mann-Whitney U test.
Cases and controls were younger and survived longer than the overall study sample, but cases and controls were similar at baseline. A moderate, statistically insignificant survival advantage was seen in the case group (Kaplan-Meier P = 0.27). However, when patients with infections in the first quarter and first half of their postoperative survival were examined, this survival advantage disappeared. There was no significant survival difference in any subgroup analyzed, including deep infections, bone flap infections, or infections caused by any specific organism.
In this single-center study, postoperative infection did not confer any survival advantage in patients with glioblastoma multiforme.
Neurosurgery 06/2009; 64(5):828-34; discussion 834-5. · 2.79 Impact Factor
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ABSTRACT: Pediatric neurosurgeons are increasingly called on to provide prognostic data regarding the antenatal diagnosis of ventriculomegaly. This study was designed to determine if there is a correlation between prenatal MR imaging results and the need for ventricular shunt placement during the neonatal period.
The authors retrospectively reviewed the prenatal MR imaging data of 38 consecutive patients who had been referred for neurosurgical consultation following the diagnosis of ventriculomegaly. The outcome measure was placement of a ventricular shunt. Assessed parameters included prenatal atrial diameter (AD), gestational age at MR imaging, time between imaging studies, presence of concomitant CNS anomalies, laterality of ventriculomegaly, fetal sex, and temporal evolution of ventriculomegaly. Logistic regression analysis was completed with the calculation of appropriate ORs and 95% CIs.
Six patients (16%) required shunt placement, all with an AD > or = 20 mm (mean 23.8 mm) at the time of imaging. Eight patients had presented with an AD > or = 20 mm. Atrial diameter was the only presenting feature that correlated with shunt placement (OR 1.58, 95% CI 1.10-2.25, p = 0.01). Logistic regression analysis revealed no statistical correlation between the need for ventricular shunting and gestational age at MR imaging, time between imaging studies, fetal sex, presence of additional CNS anomalies, and laterality of the ventriculomegaly.
When assessed using MR imaging, an AD > or = 20 mm at any gestational age is highly associated with the need for postnatal shunting. Patients with concomitant CNS anomalies did not require shunts at a greater rate than those with isolated ventriculomegaly. Further studies are required to assess the long-term outcome of this patient population.
Journal of Neurosurgery Pediatrics 05/2009; 3(5):365-70. · 1.53 Impact Factor
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ABSTRACT: Pediatric neurosurgeons frequently care for children with traumatic scalp and skull injury. Foreign objects are often observed on imaging and may influence the clinician's decision-making process. The authors report on 2 cases of poorly visualized hair beads that had become embedded into the skull during blunt trauma. In both cases, skull radiography and CT scanning demonstrated depressed, comminuted fractures with poorly demonstrated spherical radiolucencies in the overlying scalp. The nature of these objects was initially unclear, and they could have represented air that entered the scalp during trauma. In one case, scalp inspection demonstrated no evidence of the bead. In the other case, a second bead was observed at the site of scalp laceration. In both cases, the beads were surgically removed, the fractures were elevated, and the patients recovered uneventfully. Radiolucent fashion accessories, such as hair beads, may be difficult to appreciate on clinical examination and may masquerade as clinically insignificant air following cranial trauma. If they are not removed, these foreign bodies may pose the risk of an infection. Pediatric neurosurgeons should consider hair accessories in the differential diagnosis of foreign bodies that may produce skull fracture following blunt trauma.
Journal of Neurosurgery Pediatrics 01/2009; 2(6):424-6. · 1.53 Impact Factor
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ABSTRACT: Soft-tissue glomus tumors (or glomangiomas) are unrelated to neuroendocrine paragangliomas (glomus tympanicum, jugulare, and vagale). The authors present the first reported case of an orbital soft-tissue glomus tumor in a child. An 8-year-old girl developed rapidly progressive right-eye blindness, proptosis, and a sixth cranial nerve palsy. Magnetic resonance imaging demonstrated a homogeneously enhancing lesion extending from the right orbit through the superior orbital fissure to the cavernous sinus and middle cranial fossa. A biopsy specimen demonstrated the lesion to be a soft-tissue glomus tumor. Following angiography and embolization, a gross-total resection of the tumor was achieved. The patient was treated with adjuvant proton-beam radiotherapy. At 24 months follow-up her proptosis and sixth cranial nerve palsy had resolved and there was no evidence of tumor recurrence.
Journal of Neurosurgery Pediatrics 06/2008; 1(5):389-91. · 1.53 Impact Factor
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ABSTRACT: Children with sickle cell anemia (SCA) and moyamoya syndrome carry a significant risk of ischemic stroke. Given the success of encephaloduroarteriosynangiosis (EDAS) or pial synangiosis in the treatment of moyamoya disease, the purpose of this study was to examine whether it reliably and durably protected children with SCA and moyamoya syndrome against cerebrovascular complications.
The authors retrospectively reviewed a series of 12 patients with SCA who developed clinical and/or radiological evidence of moyamoya syndrome and underwent EDAS.
Eleven patients (92%) presented following a cerebrovascular accident (CVA), transient ischemic attack (TIA), or seizure. Magnetic resonance (MR) imaging or angiography suggested moyamoya vascular changes, and cerebral angiography confirmed the diagnosis in all 12 patients. At the time of surgery, the median age was 12.3 years (range 6.8-19.4 years). Ten (83%) of 12 patients had a history of CVA, and 4 of these patients were compliant with a transfusion protocol at the time of their CVA. Bilateral (7 patients) or unilateral (5 patients) EDAS was performed without complications. The mean follow-up period was 46.8 months (range 8.1-106 months). During the follow-up period, 2 patients (16.7%) suffered cerebrovascular events. One patient, who was stroke-free preoperatively, suffered a CVA 3 weeks after the procedure. The other patient suffered a single left lower-extremity TIA 18 months following right-sided EDAS. She returned to her neurological baseline condition and remains stable 53 months postoperatively. Seven patients underwent follow-up angiography or MR angiography, and evidence of revascularization was noted in all cases. At this time, no patient has developed progressive disease requiring a contralateral procedure after unilateral EDAS.
The EDAS procedure is a safe and effective treatment option in patients with SCA who develop moyamoya syndrome.
Journal of Neurosurgery Pediatrics 04/2008; 1(3):211-6. · 1.53 Impact Factor
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ABSTRACT: We sought to simulate the frontotemporal orbitozygomatic (FTOZ) craniotomy in a three-dimensional virtual environment on patient-specific data and to quantify the exposure afforded by the FTOZ while simulating controlled amounts of brain retraction.
Four computed tomographic angiograms were reconstructed with commercially available software (Amira 4.1.1; Mercury Computer Systems, Inc., Chelmsford, MA), and virtual FTOZ craniotomies were performed bilaterally (n = 8). Brain retraction was simulated at 1 and 2 cm. Surgical freedom and projection angle were measured and compared at each stage of the FTOZ.
At 1 cm of retraction, surgical freedom increased by 27 +/- 14% for the removal of the orbital rim and by 31 +/- 18% for FTOZ (P < 0.01) when compared with frontotemporal (FT) craniotomy. At 2 cm of retraction, surgical freedom increased by 15 +/- 5% and 26 +/- 8% for the removal of the orbital rim and FTOZ, respectively (P < 0.01). With increased retraction, surgical freedom increased by 100 +/- 26%, 81 +/- 15%, and 82 +/- 27% for the FT, removal of the orbital rim, and FTOZ craniotomies, respectively (P < 0.001). Projection angle increased by 24.2% when orbital rim removal was added to the FT craniotomy (P < 0.01).
Surgical freedom increases significantly at every step of the FTOZ craniotomy. This effect is less robust when brain retraction is increased. Brain retraction alone has a greater impact on surgical freedom than bone removal alone. Projection angle is significantly increased when orbital rim removal is added to the FT craniotomy. This model overcomes two major limitations of cadaver-based models: quantification of brain retraction and incorporation of patient-specific anatomy.
Neurosurgery 03/2008; 62(3 Suppl 1):251-60; discussion 260-1. · 2.79 Impact Factor
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Clinical neurosurgery 02/2008; 55:145-9.
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ABSTRACT: The authors describe the internal cranial expansion (ICE) procedure, a surgical technique that was used to treat two chronically shunt-treated children who presented with medically and surgically refractory intracranial hypertension despite the presence of functioning cerebrospinal fluid shunt systems. The ICE procedure was used as a means to increase intracranial volume without sacrificing calvarial rigidity. Intracranial volume was increased by 5% in one case and 10% in the other. Both patients have returned to their neurological and functional baselines, and they are free of symptoms related to intracranial hypertension.
Journal of Neurosurgery 12/2007; 107(5 Suppl):402-5. · 2.96 Impact Factor
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ABSTRACT: This article addresses the key features, presentation, and current management of pediatric patients who have Chiari I and Chiari II malformations. It further discusses syringohydromyelia and scoliosis as they relate to pediatric Chiari malformations.
Neurosurgery clinics of North America 08/2007; 18(3):549-68. · 1.73 Impact Factor
