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Nicholas M. Wetjen, P. Charles Garell, Nicholas V. Stence and Christopher M. Loftus

There have been few investigations of moyamoya disease in the United States and no systematic description of the management practices or outcome from this population. The authors reviewed their experience with this disease to gain a better understanding and improve the treatment of patients with moyamoya disease in the United States. Over a 25-year period 30 patients with moyamoya disease have been treated at the University of Iowa. The cases were divided into patients who had classic, probable, and akin moyamoya disease.

Results indicated that there was a bimodal age distribution and a female predominance of cases. In estimating the referral pattern of our institution, the authors determined that there were greater numbers of epidemiological characteristics than previously anticipated. Patients were treated either surgically or nonsurgically, and different management strategies were utilized in each of the major groups: superficial temporal artery to middle cerebral artery anastomosis and encephalodurosynangiosis in the surgical group; or antiplatelet, anticoagulation, or nonpharmacological intervention in the nonsurgical group.

The authors conclude that there is a higher prevalence and incidence of moyamoya disease in the United States than previously reported and that there are some clinical characteristics of this disease that differ from the cases reported in southeast Asia. These differences may be due to genetic or environmental factors but can also be partly explained by the lower index of suspicion for this disease and, thus, a delay in or complete absence of the correct diagnosis.

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Brent R. O'Neill, Danielle Gallegos, Alex Herron, Claire Palmer, Nicholas V. Stence, Todd C. Hankinson, C. Corbett Wilkinson and Michael H. Handler

OBJECTIVE

Cutaneous stigmata or congenital anomalies often prompt screening for occult spinal dysraphism (OSD) in asymptomatic infants. While a number of studies have examined the results of ultrasonography (US) screening, less is known about the findings when MRI is used as the primary imaging modality. The object of this study was to assess the results of MRI screening for OSD in infants.

METHODS

The authors undertook a retrospective review of all infants who had undergone MRI of the lumbar spine to screen for OSD over a 6-year period (September 2006–September 2012). All images had been obtained on modern MRI scanners using sequences optimized to detect OSD, which was defined as any fibrolipoma of the filum terminale (FFT), a conus medullaris ending at or below the L2–3 disc space, as well as more complex lesions such as lipomyelomeningocele (LMM).

RESULTS

Five hundred twenty-two patients with a mean age of 6.2 months at imaging were included in the study. Indications for imaging included isolated dimple in 235 patients (45%), asymmetrically deviated gluteal cleft in 43 (8%), symmetrically deviated (Y-shaped) gluteal cleft in 38 (7%), hemangioma in 28 (5%), other isolated cutaneous stigmata (subcutaneous lipoma, vestigial tail, hairy patch, and dysplastic skin) in 31 (6%), several of the above stigmata in 97 (18%), and congenital anomalies in 50 (10%).

Twenty-three percent (122 patients) of the study population had OSD. Lesions in 19% of these 122 patients were complex OSD consisting of LMM, dermal sinus tract extending to the thecal sac, and lipomeningocele. The majority of OSD lesions (99 patients [81%]) were filar abnormalities, a group including FFT and low-lying conus.

The rate of OSD ranged from 12% for patients with asymmetrically deviated gluteal crease to 55% for those with other isolated cutaneous stigmata.

Isolated midline dimple was the most common indication for imaging. Among this group, 20% (46 of 235) had OSD. There was no difference in the rate of OSD based on dimple location. Those with OSD had a mean dimple position of 15 mm (SD 11.8) above the coccyx. Those without OSD had a mean dimple position of 12.2 mm (SD 19) above the coccyx (p = 0.25).

CONCLUSIONS

The prevalence of OSD identified with modern high-resolution MRI screening is significantly higher than that reported with US screening, particularly in patients with dimples. The majority of OSD lesions identified are FFT and low conus. The clinical significance of such lesions remains unclear.

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C. Corbett Wilkinson, Nicholas V. Stence, Cesar A. Serrano, Sarah J. Graber, Lígia Batista-Silverman, Emily Schmidt-Beuchat and Brooke M. French

OBJECTIVE

Recently, the authors investigated the normal course of fusion of minor lateral calvarial sutures on “3D” volume-rendered head CT reconstructions in pediatric trauma patients. While evaluating these reconstructions, they found many more fused sagittal sutures than expected given the currently accepted prevalence of sagittal craniosynostosis. In the present study, using the same set of head CT reconstructions, they investigated the course of fusion of the sagittal as well as the lambdoid, coronal, and metopic sutures.

METHODS

They reviewed all volume-rendered head CT reconstructions performed in the period from 2010 through mid-2012 at Children’s Hospital Colorado for trauma patients aged 0–21 years. Each sagittal, lambdoid, coronal, or metopic suture was graded as open, partially fused, or fused. The cephalic index (CI) was calculated for subjects with fused and partially fused sagittal sutures.

RESULTS

After exclusions, 331 scans were reviewed. Twenty-one subjects (6%) had fusion or partial fusion of the sagittal suture. Four of the 21 also had fusion of the medial lambdoid and/or coronal sutures. In the 17 subjects (5%) with sagittal suture fusion and no medial fusion of adjacent sutures, the mean CI was 77.6. None of the 21 subjects had been previously diagnosed with craniosynostosis. Other than in the 21 subjects already mentioned, no other sagittal or lambdoid sutures were fused at all. Nor were other coronal sutures fused medially. Coronal sutures were commonly fused inferiorly early during the 2nd decade of life, and fusion progressed superiorly and medially as subjects became older; none were completely fused by 18 years of age. Fusion of the metopic suture was first seen at 3 months of life; fusion was often not complete until after 2 years.

CONCLUSIONS

The sagittal and lambdoid sutures do not usually begin to fuse before 18 years of age. However, more sagittal sutures are fused before age 18 than expected given the currently accepted prevalence of craniosynostosis. This finding is of unknown significance, but likely many of them do not need surgery. The coronal suture often begins to fuse inferiorly early in the 2nd decade of life but does not usually complete fusion before 18 years of age. The metopic suture often starts to fuse by 3 months of age, but it may not completely fuse until after 2 years of age.

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C. Corbett Wilkinson, Cesar A. Serrano, Brooke M. French, Sarah J. Graber, Emily Schmidt-Beuchat, Lígia Batista-Silverman, Noah P. Hubbell and Nicholas V. Stence

OBJECTIVE

Several years ago, the authors treated an infant with sagittal and bilateral parietomastoid suture fusion. This made them curious about the normal course of fusion of “minor” lateral sutures (sphenoparietal, squamosal, parietomastoid). Accordingly, they investigated fusion of these sutures on 3D volume-rendered head CT reconstructions in a series of pediatric trauma patients.

METHODS

The authors reviewed all volume-rendered head CT reconstructions obtained from 2010 through mid-2012 at Children’s Hospital Colorado in trauma patients aged 0–21 years. Each sphenoparietal, squamosal, and parietomastoid suture was graded as open, partially fused, or fused. In several individuals, one or more lateral sutures were fused atypically. In these patients, the cephalic index (CI) and cranial vault asymmetry index (CVAI) were calculated. In a separately reported study utilizing the same reconstructions, 21 subjects had fusion of the sagittal suture. Minor lateral sutures were assessed, including these 21 individuals, excluding them, and considering them as a separate subgroup.

RESULTS

After exclusions, 331 scans were reviewed. Typically, the earliest length of the minor lateral sutures to begin fusion was the anterior squamosal suture, often by 2 years of age. The next suture to begin fusion—and first to complete it—was the sphenoparietal. The last suture to begin and complete fusion was the parietomastoid. Six subjects (1.8%) had posterior (without anterior) fusion of one or more squamosal sutures. Six subjects (1.8%) had fusion or near-complete fusion of one squamosal and/or parietomastoid suture when the corresponding opposite suture was open or nearly open. The mean CI and CVAI values in these subjects and in age- and sex-matched controls were normal and not significantly different. No individuals had a fused parietomastoid suture with open squamosal and/or sphenoparietal sutures.

CONCLUSIONS

Fusion and partial fusion of the sphenoparietal, squamosal, and parietomastoid sutures is common in children and adolescents. It usually does not represent craniosynostosis and does not require cranial surgery. The anterior squamosal suture is often the earliest length of these sutures to fuse. Fusion then spreads anteriorly to the sphenoparietal suture and posteriorly to the parietomastoid. The sphenoparietal suture is generally the earliest minor lateral suture to complete fusion, and the parietomastoid is the last. Atypical patterns of fusion include posterior (without anterior) squamosal suture fusion and asymmetrical squamosal and/or parietomastoid suture fusion. However, these atypical fusion patterns may not lead to atypical head shapes or a need for surgery.

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Chima O. Oluigbo, C. Corbett Wilkinson, Nicholas V. Stence, Laura Z. Fenton, Sean A. McNatt and Michael H. Handler

Object

The goal of this study was to compare clinical outcomes following decompressive craniectomy performed for intracranial hypertension in children with nonaccidental, blunt cranial trauma with outcomes of decompressive craniectomy in children injured by other mechanisms.

Methods

All children in a prospectively acquired database of trauma admissions who underwent decompressive craniectomy over a 9-year span, beginning January 1, 2000, are the basis for this study. Clinical records and neuroimaging studies were systematically reviewed.

Results

Thirty-seven children met the inclusion criteria. Nonaccidental head trauma was the most common mechanism of injury (38%). The mortality rate in patients with abusive brain injury (35.7%) was significantly higher (p < 0.05) than in patients with other causes of traumatic brain injury (4.3%). Children with inflicted head injuries had a 12-fold increase in the odds of death and 3-fold increase in the odds of a poor outcome (King's Outcome Scale for Closed Head Injury score of 1, 2, or 3).

Conclusions

Children with nonaccidental blunt cranial trauma have significantly higher mortality following decompressive craniectomy than do children with other mechanisms of injury. This understanding can be interpreted to mean either that the threshold for decompression should be lower in children with nonaccidental closed head injury or that decompression is unlikely to alter the path to a fatal outcome. If decompressive craniectomy is to be effective in reducing mortality in the setting of nonaccidental blunt cranial trauma, it should be done quite early.

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Soliman Oushy, Jonathon J. Parker, Kristen Campbell, Claire Palmer, Corbett Wilkinson, Nicholas V. Stence, Michael H. Handler and David M. Mirsky

OBJECTIVE

Placement of a cerebrospinal fluid diversion device (i.e., shunt) is a routine pediatric neurosurgical procedure, often performed in the first weeks of life for treatment of congenital hydrocephalus. In the postoperative period, shunt placement may be complicated by subdural, catheter tract, parenchymal, and intraventricular hemorrhages. The authors observed a subset of infants and neonates who developed multifocal intraparenchymal hemorrhages (MIPH) following shunt placement and sought to determine any predisposing perioperative variables.

METHODS

A retrospective review of the electronic medical record at a tertiary-care children’s hospital was performed for the period 1998–2015. Inclusion criteria consisted of shunt placement, age < 30 days, and available pre- and postoperative brain imaging. The following data were collected and analyzed for each case: ventricular size ratios, laboratory values, clinical presentation, shunt and valve type, and operative timing and approach.

RESULTS

A total of 121 neonates met the inclusion criteria for the study, and 11 patients (9.1%) had MIPH following shunt placement. The preoperative frontal and occipital horn ratio (FOR) was significantly higher in the patients with MIPH than in those without (0.65 vs 0.57, p < 0.001). The change in FOR (∆FOR) after shunt placement was significantly greater in the MIPH group (0.14 vs 0.08, p = 0.04). Among neonates who developed MIPH, aqueductal stenosis was the most common etiology (45%). The type of shunt valve was associated with incidence of MIPH (p < 0.001). Preoperative clinical parameters, including head circumference, bulging fontanelle, and coagulopathy, were not significantly associated with development of MIPH.

CONCLUSIONS

MIPH represents an underrecognized complication of neonatal shunted hydrocephalus. Markers of severity of ventriculomegaly (FOR) and ventricular response to CSF diversion (∆FOR) were significantly associated with occurrence of MIPH. Choice of shunt and etiology of hydrocephalus were also significantly associated with MIPH. After adjusting for corrected age, etiology of hydrocephalus, and shunt setting, the authors found that ∆FOR after shunting was still associated with MIPH. A prospective study of MIPH prevention strategies and assessment of possible implications for patient outcomes is needed.

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Mohana Rao Patibandla, Thomas Ridder, Kathleen Dorris, Michelle R. Torok, Arthur K. Liu, Michael H. Handler, Nicholas V. Stence, Laura Z. Fenton and Todd C. Hankinson

OBJECT

Ganglioglioma (GG) is commonly recognized as a low-grade tumor located in the temporal lobe, often presenting with seizures. Most are amenable to complete resection and are associated with excellent oncological outcome. The authors encountered several GGs in various locations, which seem to have a less favorable clinical course than GGs in the temporal lobe.

METHODS

The authors performed a single-center retrospective review of all children with a histological diagnosis of GG who were treated at Children’s Hospital Colorado between 1997 and 2013. Each tumor was categorized by 2 pediatric neuroradiologists as typical or atypical based on preoperative MRI appearance. Typical lesions were cortically based, within a single cerebral lobe, well-circumscribed, and solid or mixed solid/cystic. The treatment and clinical course of each patient was analyzed.

RESULTS

Thirty-seven children were identified, with a median age at presentation of 8.2 years and median follow-up of 38.0 months. Eighteen tumors (48.6%) were typical and 19 (51.4%) were atypical. All typical lesions presented with seizures, whereas no atypical lesions did so. Sixteen (88.9%) typical lesions were located in the temporal lobe. In the atypical group, tumor location was variable, including 11 (57.9%) in the brainstem. Death during follow-up was statistically more common in the atypical group (31.6% vs 0%, p = 0.02). Gross-total resection (GTR) was achieved for 15 of 16 typical tumors (93.8%), compared with 3 atypical tumors (15.8%, p < 0.0001). Presentation with seizure or non-brainstem location were each associated with survival (p = 0.02 and 0.004, respectively). The presence of mutation in BRAF exon 15 did not differ between the 2 groups.

CONCLUSIONS

Pediatric GG with typical imaging features is associated with excellent rates of GTR and overall survival. Atypical GG is commonly encountered, less amenable to GTR, and associated with a worse outcome. This may relate to anatomical or biological characteristics and merits further investigation.