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Management of severe cervical kyphosis in a patient with Larsen syndrome

Case report

Marci Madera, Alvin Crawford, and Francesco T. Mangano

✓ Larsen syndrome is a rare genetic disorder of the connective tissue that is characterized by multiple joint dislocations, distinctive deformities of the hands and feet, characteristic facial features, kyphoscoliosis, and segmentation anomalies of the vertebrae. Diverse treatment options, including conservative observation and surgical correction, have been reported for patients who present with cervical spine pathophysiology. Differences in surgical approaches, timing of the correction, and pre- or postoperative bracing have been reported. According to the authors, the present case is the first report of a pediatric patient with Larsen syndrome in whom an asymptomatic cervical instability was treated before neurological deterioration with synchronous anterior decompression and fixation, posterior fusion and fixation, and halo placement. A review of the literature on similar patients reveals the variety of practices associated with a diagnosis of Larsen syndrome.

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Standardizing preoperative preparation to reduce surgical site infections among pediatric neurosurgical patients

Joshua K. Schaffzin, Katherine Simon, Beverly L. Connelly, and Francesco T. Mangano

OBJECTIVE

Surgical site infections (SSIs) are costly to patients and the health care system. Pediatric neurosurgery SSI risk factors are not well defined. Intraoperative protocols have reduced, but have not eliminated, SSIs. The effect of preoperative intervention is unknown. Using quality improvement methods, a preoperative SSI prevention protocol for pediatric neurosurgical patients was implemented to assess its effect on SSI rate.

METHODS

Patients who underwent a scheduled neurosurgical procedure between January 2014 and December 2015 were included. Published evidence and provider consensus were used to guide preoperative protocol development. The Model for Improvement was used to test interventions. Intraoperative and postoperative management was not standardized or modified systematically. Staff, family, and overall adherence was measured as all-or-nothing. In addition, SSI rates among eligible procedures were measured before and after protocol implementation.

RESULTS

Within 4 months, overall protocol adherence increased from 51.3% to a sustained 85.7%. SSI rates decreased from 2.9 per 100 procedures preintervention to 0.62 infections postintervention (p = 0.003). An approximate 79% reduction in SSI risk was identified (risk ratio 0.21, 95% CI 0.08–0.56; p = 0.001).

CONCLUSIONS

Clinical staff and families successfully collaborated on a standardized preoperative protocol for pediatric neurosurgical patients. Standardization of the preoperative phase of care alone reduced SSI rates. Attention to the preoperative in addition to the intraoperative and postoperative phases of care may lead to further reduction in SSI rates.

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Characterization of abnormal diffusion properties of supratentorial brain tumors: a preliminary diffusion tensor imaging study

Weihong Yuan, Scott K. Holland, Blaise V. Jones, Kerry Crone, and Francesco T. Mangano

Object

Diffusion tensor (DT) imaging was used in children with supratentorial tumors to evaluate the anisotropic diffusion properties between different tumor grades and between tumors and adjacent and contralateral white matter.

Methods

In this retrospective review, the authors review the cases of 16 children (age range 1–18 years) who presented to their institution with supratentorial tumors and were treated between 2004 and 2007. Eleven patients had low-grade and 5 had high-grade tumors. Fractional anisotropy (FA), mean diffusivity, and axial (λ) and radial (λ) eigenvalues within selected regions were studied. Mitotic index, necrosis, and vascularity of the tumors were compared with DT imaging parameters.

Results

The mean diffusivity was significantly higher in low-grade than in high-grade tumors (p = 0.04); the 2 tumor grades also significantly differed for both λ (p < 0.05) and λ (p < 0.05). Mean diffusivity values in low-grade tumors were significantly higher than in adjacent normal-appearing white matter (NAWM; p = 0.0004) and contralateral NAWM (p = 0.0001). In both low- and high-grade tumors, the FA was significantly lower than in NAWM (p < 0.0001 and p < 0.03, respectively) and contralateral NAWM (p < 0.0001 and p < 0.003, respectively). Tumor cellularity highly correlated with mean diffusivity and λand λ.

Conclusions

Diffusion tensor imaging is a useful tool in the evaluation of supratentorial tumors in children. The mean diffusivity appears to be a significant marker in differentiating tumors grades. Findings related to λ and λ within tumor groups and between tumors and NAWM may be an indirect manifestation of the combined effects of axonal injury, demyelination, and tumor mass within the cranial compartment.

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Management of vagal nerve stimulator infections: do they need to be removed?

Clinical article

Ellen L. Air, Yashar M. Ghomri, Rachana Tyagi, Andrew W. Grande, Kerry Crone, and Francesco T. Mangano

Object

Vagal nerve stimulators (VNSs) have been used successfully to treat medically refractory epilepsy. Although their efficacy is well established, appropriate management of infections is less clearly defined. In the authors' experience, patients who have gained a benefit from VNS implantation have been reluctant to have the device removed. The authors therefore sought conservative management options to salvage infected VNS systems.

Methods

The authors performed a retrospective review of 191 (93 female and 98 male) consecutive patients in whom VNS systems were placed between 2000 and 2007.

Results

They identified 10 infections (5.2%). In 9 of 10 patients the cultured organism was Staphylococcus aureus. Three (30%) of 10 patients underwent early removal (within 1 month) of the VNS as the initial treatment. The remaining 7 patients were initially treated with antibiotics. Two (28.6%) of these patients were successfully treated using antibiotics without VNS removal. Patients in whom conservative treatment failed were given cephalexin as first-line antibiotic treatment. All patients recovered completely regardless of treatment regimen.

Conclusions

This study confirms the low rate of infection associated with VNS placement and suggests that, in the case of infection, treatment without removal is a viable option. However, the authors' data suggest that oral antibiotics are not the best first-line therapy.

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Mesial temporal lobe morphology in intractable pediatric epilepsy: so-called hippocampal malrotation, associated findings, and relevance to presurgical assessment

James L. Leach, Reem Awwad, Hansel M. Greiner, Jennifer J. Vannest, Lili Miles, and Francesco T. Mangano

OBJECTIVE

Diagnostic criteria for hippocampal malrotation (HIMAL) on brain MRI typically include a rounded hippocampus, vertical collateral sulcus, and architectural blurring. Relationship to epileptogenesis remains speculative, and usefulness for surgical guidance is unknown. The study was performed to determine the prevalence of hippocampal rotational anomalies in a cohort of pediatric patients with intractable epilepsy undergoing evaluation for surgery and to determine the significance of this finding in the context of surgical planning.

METHODS

Forty-eight surgically treated children with intractable epilepsy were compared with matched healthy subjects; reviewers were blinded to surgical side. Each temporal lobe was evaluated for rounded hippocampus, blurring, vertical collateral sulcus, wide choroidal fissure, enlarged temporal horn, low fornix, hippocampal signal, and findings of hippocampal sclerosis. A mesial temporal lobe (MTL) score was calculated by summing the number of features, and the collateral sulcus angle (CSA) was measured in each temporal lobe. Surgical side, pathological diagnosis, and imaging findings elsewhere in the brain were tabulated. Presence of HIMAL, associated imaging features, and MTL score were compared between sides, between epilepsy and control groups, in relationship to side of surgery, and in relationship to postoperative outcome.

RESULTS

Only 3 epilepsy patients (6.2%) and no controls exhibited all 3 features of HIMAL (p = 0.12). Eight of 48 (16.7%) epilepsy versus 2 of 48 (4.6%) control subjects had both a rounded hippocampus and vertical collateral sulcus (suggesting HIMAL) (p = 0.045). In control and epilepsy subjects, most findings were more prevalent on the left, and the left CSA was more vertical (p < 0.0001). Epilepsy subjects had higher MTL scores (z = −2.95, p = 0.002) and more acute CSAs (p = 0.04) than controls. Only lateralizing raw MTL score had a significant association with surgical side (p = 0.03, OR 7.33); however, this was not significant when hippocampal sclerosis cases were excluded. HIMAL findings were more prevalent and MTL scores were higher in patients with resections involving the temporal lobes. On group analysis, HIMAL findings did not predict eventual surgical side and did not predict outcome, although the numbers are small. In 4 patients the abnormally rotated hippocampus was resected and showed hippocampal sclerosis and/or dysplastic changes on histopathology. All of these patients had a good outcome after surgery.

CONCLUSIONS

While increased in prevalence in children with intractable epilepsy, imaging findings of HIMAL did not have preoperative lateralizing utility in this group. Findings of HIMAL (including round hippocampus, architectural blurring, and vertical collateral sulcus) did not predict outcome after surgery, although the small number of patients with these findings limits evaluation. In the small number of patients in which the malrotated hippocampus was removed, outcome was good. Further research is needed to continue to define this association in children with intractable epilepsy, focusing on a temporal lobe cohort.

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Categorizing cortical dysplasia lesions for surgical outcome using network functional connectivity

Abdullah S. Bdaiwi, Hansel M. Greiner, James Leach, Francesco T. Mangano, and Mark W. DiFrancesco

OBJECTIVE

Focal cortical dysplasia (FCD) is often associated with drug-resistant epilepsy, leading to a recommendation to surgically remove the seizure focus. Predicting outcome for resection of FCD is challenging, requiring a new approach. Lesion-symptom mapping is a powerful and broadly applicable method for linking neurological symptoms or outcomes to damage to particular brain regions. In this work, the authors applied lesion network mapping, an expansion of the traditional approach, to search for the association of lesion network connectivity with surgical outcomes. They hypothesized that connectivity of lesion volumes, preoperatively identified by MRI, would associate with seizure outcomes after surgery in a pediatric cohort with FCD.

METHODS

This retrospective study included 21 patients spanning the ages of 3 months to 17.7 years with FCD lesions who underwent surgery for drug-resistant epilepsy. The mean brain-wide functional connectivity map of each lesion volume was assessed across a database of resting-state functional MRI data from healthy children (spanning approximately 2.9 to 18.9 years old) compiled at the authors’ institution. Lesion connectivity maps were averaged across age and sex groupings from the database and matched to each patient. The authors sought to associate voxel-wise differences in these maps with subject-specific surgical outcome (seizure free vs persistent seizures).

RESULTS

Lesion volumes with persistent seizures after surgery tended to have stronger connectivity to attention and motor networks and weaker connectivity to the default mode network compared with lesion volumes with seizure-free surgical outcome.

CONCLUSIONS

Network connectivity–based lesion-outcome mapping may offer new insight for determining the impact of lesion volumes discerned according to both size and specific location. The results of this pilot study could be validated with a larger set of data, with the ultimate goal of allowing examination of lesions in patients with FCD and predicting their surgical outcomes.

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Longitudinal comparison of pre- and postoperative diffusion tensor imaging parameters in young children with hydrocephalus

Clinical article

Ellen L. Air, Weihong Yuan, Scott K. Holland, Blaise V. Jones, Karin Bierbrauer, Mekibib Altaye, and Francesco T. Mangano

Object

The goal in this study was to compare the integrity of white matter before and after ventriculoperitoneal (VP) shunt insertion by evaluating the anisotropic diffusion properties with the aid of diffusion tensor (DT) imaging in young children with hydrocephalus.

Methods

The authors retrospectively identified 10 children with hydrocephalus who underwent both pre- and postoperative DT imaging studies. The DT imaging parameters (fractional anisotropy [FA], mean diffusivity, axial diffusivity, and radial diffusivity) were computed and compared longitudinally in the splenium and genu of the corpus callosum (gCC) and in the anterior and posterior limbs of the internal capsule (PLIC). The patients' values on DT imaging at the pre- and postshunt stages were compared with the corresponding age-matched controls as well as with a large cohort of healthy children in the database.

Results

In the gCC, 7 of 10 children had abnormally low preoperative FA values, 6 of which normalized postoperatively. All 3 of the 10 children who had normal preoperative FA values had normal FA values postoperatively as well. In the PLIC, 7 of 10 children had abnormally high FA values, 6 of which normalized postoperatively, whereas the other one had abnormally low postoperative FA. Of the remaining 3 children, 2 had abnormally low preoperative FA values in the PLIC; this normalized in 1 patient after surgery. The other child had a normal preoperative FA value that became abnormally low postoperatively. When comparing the presurgery frequency of abnormally low, normal, and abnormally high FA values to those postsurgery, there was a statistically significant longitudinal difference in both gCC (p = 0.02) and PLIC (p = 0.002).

Conclusions

In this first longitudinal DT imaging study of young children with hydrocephalus, DT imaging anisotropy yielded abnormal results in several white matter regions of the brain, and trended toward normalization following VP shunt placement.

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Diffusion tensor imaging in children following prenatal myelomeningocele repair and its predictive value for the need and timing of subsequent CSF diversion surgery for hydrocephalus

Weihong Yuan, Charles B. Stevenson, Mekibib Altaye, Blaise V. Jones, James Leach, Mykhailo Lovha, Noa Rennert, and Francesco T. Mangano

OBJECTIVE

The aim of this study was to investigate diffusion tensor imaging (DTI), an objective and noninvasive neuroimaging technique, for its potential as an imaging biomarker to predict the need and timing of CSF diversion surgery in patients after prenatal myelomeningocele (MMC) repair.

METHODS

This was a retrospective analysis of data based on 35 pediatric patients after prenatal MMC repair (gestational age at birth 32.68 ± 3.42 weeks, range 24–38 weeks; 15 females and 20 males). A logistic regression analysis was used to classify patients to determine the need for CSF diversion surgery. The model performance was compared between using the frontooccipital horn ratio (FOHR) alone and using the FOHR combined with DTI values (the genu of the corpus callosum [gCC] and the posterior limb of the internal capsule [PLIC]). For patients who needed to be treated surgically, timing of the procedure was used as the clinical outcome to test the predictive value of DTI acquired prior to surgery based on a linear regression analysis.

RESULTS

Significantly lower fractional anisotropy (FA) values in the gCC (p = 0.014) and PLIC (p = 0.037) and higher mean diffusivity (MD) values in the gCC (p = 0.013) were found in patients who required CSF diversion surgery compared with those who did not require surgery (all p values adjusted for age). Based on the logistic regression analysis, the FOHR alone showed an accuracy of performance of 0.69 and area under the receiver operating characteristic curve (AUC) of 0.60. The performance of the model was higher when DTI measures were used in the logistic regression model (accuracy = 0.77, AUC = 0.84 for using DTI values in gCC; accuracy = 0.75, AUC = 0.84 for using DTI values in PLIC). Combining the DTI values of the gCC or PLIC and FOHR did not improve the model performance when compared with using the DTI values alone. In patients who needed CSF diversion surgery, significant correlation was found between DTI values in the gCC and the time interval between imaging and surgery (FA: ρ = 0.625, p = 0.022; MD: ρ = −0.6830, p = 0.010; both adjusted for age and FOHR).

CONCLUSIONS

The authors’ data demonstrated that DTI could potentially serve as an objective biomarker differentiating patients after prenatal MMC repair regarding those who may require surgery for MMC-associated hydrocephalus. The predictive value for the need and timing of CSF diversion surgery is highly clinically relevant for improving and optimizing decision-making for the treatment of hydrocephalus in this patient population.

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Neonatal hydrocephalus leads to white matter neuroinflammation and injury in the corpus callosum of Ccdc39 hydrocephalic mice

Danielle S. Goulding, R. Caleb Vogel, Chirayu D. Pandya, Crystal Shula, John C. Gensel, Francesco T. Mangano, June Goto, and Brandon A. Miller

OBJECTIVE

The authors sought to determine if hydrocephalus caused a proinflammatory state within white matter as is seen in many other forms of neonatal brain injury. Common causes of hydrocephalus (such as trauma, infection, and hemorrhage) are inflammatory insults themselves and therefore confound understanding of how hydrocephalus itself affects neuroinflammation. Recently, a novel animal model of hydrocephalus due to a genetic mutation in the Ccdc39 gene has been developed in mice. In this model, ciliary dysfunction leads to early-onset ventriculomegaly, astrogliosis, and reduced myelination. Because this model of hydrocephalus is not caused by an antecedent proinflammatory insult, it was utilized to study the effect of hydrocephalus on inflammation within the white matter of the corpus callosum.

METHODS

A Meso Scale Discovery assay was used to measure levels of proinflammatory cytokines in whole brain from animals with and without hydrocephalus. Immunohistochemistry was used to measure macrophage activation and NG2 expression within the white matter of the corpus callosum in animals with and without hydrocephalus.

RESULTS

In this model of hydrocephalus, levels of cytokines throughout the brain revealed a more robust increase in classic proinflammatory cytokines (interleukin [IL]–1β, CXCL1) than in immunomodulatory cytokines (IL-10). Increased numbers of macrophages were found within the corpus callosum. These macrophages were polarized toward a proinflammatory phenotype as assessed by higher levels of CD86, a marker of proinflammatory macrophages, compared to CD206, a marker for antiinflammatory macrophages. There was extensive structural damage to the corpus callosum of animals with hydrocephalus, and an increase in NG2-positive cells.

CONCLUSIONS

Hydrocephalus without an antecedent proinflammatory insult induces inflammation and tissue injury in white matter. Future studies with this model will be useful to better understand the effects of hydrocephalus on neuroinflammation and progenitor cell development. Antiinflammatory therapy for diseases that cause hydrocephalus may be a powerful strategy to reduce tissue damage.

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Longitudinal comparison of diffusion tensor imaging parameters and neuropsychological measures following endoscopic third ventriculostomy for hydrocephalus

Case report

Robert T. Buckley, Weihong Yuan, Francesco T. Mangano, Jannel M. Phillips, Stephanie Powell, Robert C. McKinstry, Akila Rajagopal, Blaise V. Jones, Scott Holland, and David D. Limbrick Jr.

The authors report the case of a 25-month-old boy who underwent endoscopic third ventriculostomy (ETV) for hydrocephalus resulting from aqueductal stenosis. The patient's recovery was monitored longitudinally and prospectively using MR diffusion tensor imaging (DTI) and formal neuropsychological testing. Despite minimal change in ventricle size, improvement in the DTI characteristics and neurodevelopmental trajectory was observed following ETV. These data support the use of DTI as a biomarker to assess therapeutic response in children undergoing surgical treatment for hydrocephalus. In the patient featured in this report, DTI appeared to provide more information regarding postoperative neurodevelopmental outcome than ventricle size alone.