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  • Journal of Neurosurgery: Pediatrics x
  • By Author: Nanda, Anil x
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Subhas K. Konar, Shyamal C. Bir, Tanmoy K. Maiti and Anil Nanda

OBJECTIVE

The incidence of primary spinal cord glioblastoma multiforme (GBM) in the pediatric age group is very rare. Only a few case series and case reports have been published in the literature; therefore, overall survival (OS) outcome and the as-yet poorly defined management options are not discussed in detail. The authors performed a cumulative survival analysis of all reported cases of pediatric spinal cord GBM to identify the predictive factors related to final survival outcome.

METHODS

A comprehensive search for relevant articles was performed on PubMed's electronic database MEDLINE for the period from 1950 to 2015 using the search words “malignant spinal cord tumor” and “spinal glioblastoma multiforme.” This study was limited to patients younger than 18 years of age. Survival rates for children with various tumor locations and treatments were collected from the published articles and analyzed.

RESULTS

After an extensive literature search, 29 articles met the study inclusion criteria. From the detailed information in these articles, the authors found 53 children eligible for the survival analysis. The majority (45%) of the children were more than 12 years old. Thirty-four percent of the cases were between 7 and 12 years of age, and 21% were younger than 7 years. In the Kaplan-Meier survival analysis, children younger than 7 years of age had better survival (13 months) than the children older than 7 years (7–12 years: 10 months, > 12 years: 9 months; p = 0.01, log-rank test). Fifty-five percent of the children were female and 45% were male. A cervical tumor location (32%) was the most common, followed by thoracic (28.3%). Cervicothoracic (18.9%) and conus (18.8%) tumor locations shared the same percentage of cases. Cervical tumors had a worse outcome than tumors in other locations (p = 0.003, log-rank test). The most common presenting symptom was limb weakness (53%), followed by sensory disturbances (25%). Median OS was 10 months. The addition of adjuvant therapy (radiotherapy [RT] and/or chemotherapy [CT]) after surgery significantly improved OS (p = 0.01, log-rank test). Children who underwent gross-total resection and RT had better outcomes than those who underwent subtotal resection and RT (p = 0.04, log-rank test). Cerebrospinal fluid spread, hydrocephalus, brain metastasis, and spinal metastasis were not correlated with OS in primary spinal GBM.

CONCLUSIONS

Adjuvant therapy after surgery had a beneficial effect on overall outcome of spinal GBM in the pediatric age group. Gross-total resection followed by RT produced a better outcome than subtotal resection with RT. Further large-scale prospective study is required to establish the genetic and molecular factors related to OS in primary GBM of the spinal cord in pediatric patients.

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Shyamal C. Bir, Piyush Kalakoti, Christina Notarianni and Anil Nanda

In the late 18th and early 19th centuries, Dr. John Howship, a pioneering British surgeon, described the clinical features and pathophysiology of various surgical disorders of the human body. His critical contributions to pediatric neurosurgery came in 1816 when he first described the features of an important childhood condition following head trauma, what he referred to as parietal bone absorption. This condition as depicted by Dr. Howship was soon to be christened by later scholars as traumatic cephalhydrocele, traumatic meningocele, leptomeningeal cyst, meningocele spuria, fibrosing osteitis, cerebrocranial erosion, and growing skull fracture. Nevertheless, the basic features of the condition as observed by Dr. Howship were virtually identical to the characteristics of the above-mentioned disorders. This article describes the life and accomplishments of Dr. Howship and his contributions to the current understanding of growing skull fracture.

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Sunil Kukreja, Sudheer Ambekar, Anthony Hunkyun Sin and Anil Nanda

Object

Reports of myxopapillary ependymomas (MPEs) of the spinal cord in pediatric patients are scarce. In the literature, various authors have shared their experiences with small groups of patients, which makes it difficult to create a consensus regarding the treatment approach for spinal MPEs in young patients. The aim of this study was to perform a survival analysis of patients in the first 2 decades of life whose cases were selected from the published studies, and to examine the influence of various factors on outcomes.

Methods

A comprehensive search of studies published in English was performed on PubMed. Patients whose age was ≤ 20 years were included for integrative analysis. Information about age, treatment characteristics, critical events (progression, recurrence, and death), time to critical events, and follow-up duration was recorded. The degree of association of the various factors with the survival outcome was calculated by using Kaplan-Meier estimator and Cox proportional hazard model techniques.

Results

A total of 95 patients were included in the analysis. The overall rate of recurrence (RR) was 34.7% (n = 33), with a median time to recurrence of 36 months (range 2–100 months). Progression-free survival (PFS) and overall survival rates at 5 years were 73.7% and 98.9%, respectively. Addition of radiotherapy (RT) following resection significantly improved PFS (log-rank test, p = 0.008). In patients who underwent subtotal resection (STR), administering RT (STR + RT) improved outcome with the lowest failure rates (10.3%), superior to patients who underwent gross-total resection (GTR) alone (RR 43.1%; log-rank test, p < 0.001). Addition of RT to patients who underwent GTR was not beneficial (log-rank test, p = 0.628). In patients who had disseminated tumor at presentation, adjuvant RT controlled the disease effectively. High-dose RT (≥ 50 Gy) did not change PFS (log-rank test, p = 0.710).

Conclusions

Routine inclusion of RT in the treatment protocol for spinal MPEs in young patients should be considered. Complete resection is always the goal of tumor resection. However, when complete resection does not seem to be possible in complex lesions, RT should be used as an adjunct to avoid aggressive resection and to minimize inadvertent injury to the surrounding neural tissues. High-dose RT (≥ 50 Gy) did not provide additional survival benefits, although this association needs to be evaluated by prospective studies.

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Prashant Chittiboina, Helena Pasieka, Ashish Sonig, Papireddy Bollam, Christina Notarianni, Brian K. Willis and Anil Nanda

Object

Cerebrospinal fluid shunts in patients with posthemorrhagic hydrocephalus are prone to failure, with some patients at risk for multiple failures. The objective of this study was to identify factors leading to multiple failures.

Methods

The authors performed a retrospective analysis of cases of posthemorrhagic hydrocephalus requiring neurosurgical intervention between 1982 and 2010.

Results

In the 109 cases analyzed, 54% of the patients were male, their mean birth weight was 1223 g, and their mean head circumference 25.75 cm. The mean duration of follow-up was 6 years, and 9 patients died. Grade III intraventricular hemorrhage was seen in 47.7% and Grade IV in 43.1%. Initial use of a ventricular access device was needed in 65 patients (59.6%), but permanent CSF shunting was needed in 104 (95.4%). A total of 454 surgical procedures were performed, including 304 shunt revisions in 78 patients (71.6%). Detailed surgical notes were available for 261 of these procedures, and of these, 51% were proximal revisions, 13% distal revisions, and 17% total shunt revisions. Revision rates were not affected by catheter type, patient sex, presence of congenital anomalies, or type of hydrocephalus. Age of less than 30 days at the initial procedure was associated with decreased survival of the first shunt. Regression analysis revealed that lower estimated gestational age (EGA) and obstructive hydrocephalus were significant predictors of multiple shunt revisions.

Conclusions

We found a high rate of need for permanent CSF shunts (95.4%) in patients with posthemorrhagic hydrocephalus. Shunt revision was required in 71.6% of patients, with those with lower birth weight and EGA at a higher risk for revisions.

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Christina Notarianni, Prasad Vannemreddy, Gloria Caldito, Papireddy Bollam, Esther Wylen, Brian Willis and Anil Nanda

Object

Hydrocephalus is a notorious neurosurgical disease that carries the adage “once a shunt always a shunt.” This study was conducted to review the treatment results of pediatric hydrocephalus.

Methods

Pediatric patients who underwent ventriculoperitoneal shunt surgery over the past 14 years were reviewed for shunt revisions. Variables studied included age at shunt placement, revision, or replacement; programmable shunts; infection; obstruction; and diagnosis (congenital, posthemorrhagic, craniospinal dysraphism, and others including trauma, tumors, and infection). Multiple regression analysis methods were used to determine independent risk factors for shunt failure and the number of shunt revisions. The Kaplan-Meier method of survival analysis was used to compare etiologies on the 5-year survival (revision-free) rate and the median 5-year survival time.

Results

A total of 253 patients were studied with an almost equal sex distribution. There were 92 patients with congenital hydrocephalus, 69 with posthemorrhagic hydrocephalus, 48 with craniospinal dysraphism, and 44 with other causes. Programmable shunts were used in 73 patients (other types of shunts were used in 180 patients). A total of 197 patients (78%) underwent revision surgeries due to shunt failures. The mortality rate was 1.6%. Age at first revision, the 5-year survival rate, and the median 5-year survival time were significantly less for both posthemorrhagic and craniospinal dysraphism than for either the congenital or “other” group (p < 0.05). The failure rate and number of revisions were not significantly reduced with programmable shunts compared with either pressure-controlled or no-valve shunts (p > 0.5).

Conclusions

Posthemorrhagic hydrocephalus and craniospinal dysraphism hydrocephalus had significantly earlier revisions than congenital and other etiologies. Programmable systems did not reduce the failure rate or the average number of shunts revisions.

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Brian Willis, Vijayakumar Javalkar, Prasad Vannemreddy, Gloria Caldito, Junko Matsuyama, Bharat Guthikonda, Papireddy Bollam and Anil Nanda

Object

The aim of the study was to analyze the outcome of surgical treatment for posthemorrhagic hydrocephalus in premature infants.

Methods

From 1990 to 2006, 32 premature infants underwent surgical treatment for posthemorrhagic hydrocephalus, and their charts were retrospectively reviewed to analyze the complications and outcome with respect to shunt revisions. Multivariate analysis and time series were used to identify factors that influence the outcome in terms of shunt revisions.

Results

The mean gestational age was 27 ± 3.3 weeks, and mean birth weight was 1192 ± 660 g. Temporary reservoir placement was performed in 15 patients, while 17 underwent permanent CSF diversion with a ventriculoperitoneal (VP) shunt. In 2 patients, reservoir tapping alone was sufficient to halt the progression of hydrocephalus; 29 patients received VP shunts. The mean follow-up period was 37.3 months. The neonates who received VP shunts first were significantly older (p = 0.02) and heavier (p = 0.04) than those who initially underwent reservoir placement. Shunts were revised in 14 patients; 42% of patients in the reservoir group had their shunts revised, while 53% of infants who had initially received a VP shunt required a revision. The revision rate per patient in the reservoir group was half that in the direct VP shunt group (p = 0.027). No patient in the reservoir group had > 2 revisions. Shunt infections developed in 3 patients (10.3%), and 2 patients in the reservoir group died of nonneurological issues related to prematurity.

Conclusions

Birth weight and age are useful parameters in decision making. Preterm neonates with low birth weights benefit from initial CSF drainage procedures followed by permanent CSF diversion with respect to the number of shunt revisions.