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Editorial

Prediction, with restriction

Alan R. Cohen

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Editorial

Shunt failure

James M. Drake

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Abhaya V. Kulkarni, James M. Drake, John R. W. Kestle, Conor L. Mallucci, Spyros Sgouros and Shlomi Constantini

Object

The authors recently developed and internally validated the ETV Success Score (ETVSS)—a simplified means of predicting the 6-month success rate of endoscopic third ventriculostomy (ETV) for a child with hydrocephalus, based on age, etiology of hydrocephalus, and presence of a previous shunt. A high ETVSS predicts a high chance of early ETV success. In this paper, they assess the clinical utility of the ETVSS by determining whether long-term survival outcomes for ETV versus shunt insertion are different within strata of ETVSS (low, moderate, and high scores).

Methods

A multicenter, international cohort of children (≤ 19 years old) with newly diagnosed hydrocephalus treated with either ETV (489 patients) or shunt insertion (720 patients) was analyzed. The ETVSS was calculated for all patients. Survival analyses with time-dependent modeling of the hazard ratios were performed.

Results

For the High-ETVSS Group (255 ETV-treated patients, 117 shunt-treated patients), ETV appeared to have a lower risk of failure right from the early postoperative phase and became more favorable with time. For the Moderate-ETVSS Group (172 ETV-treated patients, 245 shunt-treated patients), ETV appeared to have a higher initial failure rate, but after about 3 months the instantaneous risk of ETV failure became slightly lower than shunt failure (that is, the hazard ratio became < 1). For the Low-ETVSS Group (62 ETV-treated patients, 358 shunt-treated patients), the early risk of ETV failure was much higher than the risk of shunt failure, but the instantaneous risk of ETV failure became lower than the risk of shunt failure at about 6 months following surgery (the hazard ratio became < 1).

Conclusions

Across all ETVSS strata, the risk of ETV failure becomes progressively lower compared with the risk of shunt failure with increasing time from the surgery. In the best ETV candidates (ETVSS ≥ 80), however, the risk of ETV failure is lower than the risk of shunt failure very soon after surgery, while for less-than-ideal ETV candidates (ETVSS ≤ 70), the risk of ETV failure is initially higher than the risk of shunt failure and only becomes lower after 3–6 months from surgery. These results need to be confirmed by larger, prospective, and preferably randomized studies.

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James M. Drake, Jay Riva-Cambrin, Andrew Jea, Kurtis Auguste, Mandeep Tamber and Maria Lamberti-Pasculli

Object

Complications of specific pediatric neurosurgical procedures are well recognized. However, focused surveillance on a specific neurosurgical unit, for all procedures, may lead to better understanding of the most important complications, and allow targeted strategies for quality improvement.

Methods

The authors prospectively recorded the morbidity and mortality events at a large pediatric neurosurgical unit over a 2-year period. Morbidity was defined as any significant adverse outcome or death (for obstructive shunt failure, within 30 days). Multiple and unrelated complications in the same patient were recorded as separate events.

Results

There were 1082 surgical procedures performed during the evaluation period. One hundred seventy-seven complications (16.4%) occurred in 147 patients. By procedure, the most common complications occurred in vascular surgery (41.7%) and brain tumor surgery (27.9%). The most common complications were CSF leakage (31 cases), a new neurological deficit (27 cases), early shunt or endoscopic third ventriculostomy obstruction (27 cases), and shunt infection (24 cases). Meningitis occurred in 19 cases: in 58% of shunt infections, 13% of CSF leaks, and 10% of wound infections. Sixty-four percent of adverse events required a second procedure, most commonly an external ventricular drain placement or shunt revision.

Conclusions

Complications in pediatric neurosurgical procedures are common, result in significant morbidity, and more than half the time require a repeat surgical procedure. Targeted strategies to prevent common complications, such as shunt infections or CSF leaks, might significantly reduce this burden.

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James A. J. King, Kurtis I. Auguste, William Halliday, James M. Drake and Abhaya V. Kulkarni

Hydrocephalus secondary to giant retrocerebellar cysts in infancy is a challenging condition and many treatment options exist. The authors report on 3 consecutive cases involving infants under the age of 6 months treated successfully with ventriculocystostomy in combination with direct hydrocephalus treatment (endoscopic third ventriculostomy or shunt placement). They describe the operative procedure, the surgical morbidity, and outcome in each case and review the literature regarding surgical approaches to this condition.

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Sasha C. Burn, Reinhard Zeller and James M. Drake

Object

Intrathecal baclofen is an effective treatment for spasticity in patients with cerebral palsy. There has been increasing concern, however, that intrathecal baclofen may accelerate the development of scoliosis in this population. To this end, the authors reviewed their population of pediatric patients with baclofen pumps to assess the incidence of scoliosis.

Methods

This was a retrospective chart and radiology review of all pediatric patients with baclofen pumps. Cobb angles were measured preoperatively and on follow-up images.

Results

Of 38 patients identified, 32 had adequate data available for inclusion in the study (16 with cerebral palsy, 7 with dystonic cerebral palsy, 4 with head injury, and 5 with other diagnoses). The mean age at pump insertion was 10.6 years and the mean follow-up period was 31 months (range 1–118 months). The mean annual Cobb angle progression was 19° (range 0–68°, median 12°).

Conclusions

In the authors' group of patients there was notable development and progression of scoliosis at a greater than previously reported rate for the same patient population, and also greater than previously reported patients with intrathecal baclofen pumps. The largest possible confounding factor in this study was the insertion of the pump before skeletal maturity and therefore coinciding with the time when scoliosis may be developing naturally. A prospective study is recommended to gather further data on the development of scoliosis in this particular population with intrathecal baclofen pumps.

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Ichiro Sugiyama, Katsumi Imai, Yu Yamaguchi, Ayako Ochi, Yoko Akizuki, Cristina Go, Tomoyuki Akiyama, O. Carter Snead III, James T. Rutka, James M. Drake, Elysa Widjaja, Sylvester H. Chuang, Doug Cheyne and Hiroshi Otsubo

Object

Magnetoencephalography (MEG) has been typically used to localize epileptic activity by modeling interictal activity as equivalent current dipoles (ECDs). Synthetic aperture magnetometry (SAM) is a recently developed adaptive spatial filtering algorithm for MEG that provides some advantages over the ECD approach. The SAM-kurtosis algorithm (also known as SAM[g2]) additionally provides automated temporal detection of spike sources by using excess kurtosis value (steepness of epileptic spike on virtual sensors). To evaluate the efficacy of the SAM(g2) method, the authors applied it to readings obtained in children with intractable epilepsy secondary to tuberous sclerosis complex (TSC), and compared them to localizations obtained with ECDs.

Methods

The authors studied 13 children with TSC (7 girls) whose ages ranged from 13 months to 16.3 years (mean 7.3 years). Video electroencephalography, MR imaging, and MEG studies were analyzed. A single ECD model was applied to localize ECD clusters. The SAM(g2) value was calculated at each SAM(g2) virtual voxel in the patient's MR imaging–defined brain volume. The authors defined the epileptic voxels of SAM(g2) (evSAM[g2]) as those with local peak kurtosis values higher than half of the maximum. A clustering of ECDs had to contain ≥ 6 ECDs within 1 cm of each other, and a grouping of evSAM(g2)s had to contain ≥ 3 evSAM(g2)s within 1 cm of each other. The authors then compared both ECD clusters and evSAM(g2) groups with the resection area and correlated these data with seizure outcome.

Results

Seizures started when patients were between 6 weeks and 8 years of age (median 6 months), and became intractable secondary to multiple tubers in all cases. Ictal onset on scalp video electroencephalography was lateralized in 8 patients (62%). The MEG studies showed multiple ECD clusters in 7 patients (54%). The SAM(g2) method showed multiple groups of epileptic voxels in 8 patients (62%). Colocalization of grouped evSAM(g2) with ECD clusters ranged from 20 to 100%, with a mean of 82%. Eight patients underwent resection of single (1 patient) and multiple (7 patients) lobes, with 6 patients achieving freedom from seizures. Of 8 patients who underwent surgery, in 7 the resection area covered ECD clusters and grouped evSAM(g2)s. In the remaining patient the resection area partially included the ECD cluster and grouped evSAM(g2)s. Six of the 7 patients became seizure free.

Conclusions

The combination of SAM(g2) and ECD analyses succeeded in localizing the complex epileptic zones in children with TSC who had intractable epilepsy secondary to multiple cortical tubers. For the subset of children with TSC who present with early-onset and nonlateralized seizures, MEG studies in which SAM(g2) and ECD are used might identify suitable candidates for resection to control seizures.

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Sasha C. Burn, Olaf Ansorge, Reinhard Zeller and James M. Drake

Object

Osteoid osteomas and osteoblastoma of the spine are rare lesions in childhood, and management strategies have changed. The authors reviewed their recent experience with these 2 types of lesions to elucidate current treatment modalities and outcomes.

Methods

Case records and radiographic images from all cases of osteoid osteoma and osteoblastoma diagnosed between 1993 and 2008 were retrospectively reviewed, including those managed nonsurgically.

Results

Thirty cases were identified; 22 were treated surgically and 8 were managed nonsurgically. The patients' mean age at presentation was 13 years (range 3–17 years). Of 30 patients, 29 (97%) presented with pain; 7 (23%) had scoliosis at presentation; 12 (40%) experienced relief with nonsteroidal antiinflammatory medication. Osteoid osteoma was diagnosed in 7 (32%) of the 22 patients who underwent surgery, and osteoblastoma in 15 (68%). Nine (41%) of the 22 surgically treated patients underwent fusion procedures (bone onlay or instrumentation) at the time of surgery. Pain freedom without medication had been achieved in 16 (73%) of the 22 surgically treated patients at a mean follow-up of 28 months (range 2–75 months) and was confirmed in 3 (38%) of the 8 nonsurgically treated patients at a mean follow-up of 33 months (range 24–51 months).

Conclusions

Osteoid osteomas and osteoblastomas can present challenging management problems in pediatric patients. In the majority of cases in which conservative therapy fails or pathological diagnosis is required, surgery using modern intraoperative imaging and spinal instrumentation can provide symptom relief and tumor control.

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Ratan D. Bhardwaj, Kurtis I. Auguste, Abhaya V. Kulkarni, Peter B. Dirks, James M. Drake and James T. Rutka

Object

Because of their location and biological behavior, brainstem cavernous malformations (CMs) pose a formidable clinical challenge to the neurosurgeon. The optimal management of these lesions requires considerable neurosurgical judgment. Accordingly, the authors reviewed their experience with the management of pediatric brainstem CMs at the Hospital for Sick Children.

Methods

The authors performed a retrospective chart review of pediatric patients who had received diagnoses of a brainstem CM at the Hospital for Sick Children over the past 20 years.

Results

Twenty patients were diagnosed with brainstem CMs. The mean age at diagnosis was 10.1 ± 5.4 years, and the patients included 13 boys and 7 girls. The mean maximal diameter of the CM was 14.3 ± 11.2 mm. The lesions were evenly distributed on the right and left sides of the brainstem with 4 midbrain, 13 pontine, and 3 medullary lesions. Seven patients underwent surgery for the management of their CMs, with a mean age at presentation of 5.2 years, and a mean CM size of 21.0 mm. Of note from the surgical group, 2 patients had a family history of CMs, 2 lesions were medullary, the CM reached a pial surface in 6 of 7 patients, and 6 of 7 lesions were located on the right side. The mean age at presentation among the 13 patients in the nonsurgical group was 12.7 years, and the mean CM size was 10.6 mm. Seven of these patients had a prior history of radiation for tumor, and only 3 had lesions that reached a pial surface.

Conclusions

The management of brainstem CMs in children is influenced by multiple factors. The majority of patients received conservative management and tended to be asymptomatic with smaller lesions. Patients with larger lesions and direct pial contact, in whom symptoms arose at a younger age were more likely to undergo surgical management. A history of familial CM was also a predictor for receiving surgical treatment. No patients with a prior history of radiation therapy underwent surgery for CMs. The presence of multiple lesions seemed to have no impact on the type of management chosen. Patients who underwent surgery did suffer morbidity related to the procedure, and tended to improve clinically over time. Conservative management was associated with new deficits arising in children, some of which improved with time. Consideration of many clinical and radiological parameters is thus prudent when managing the care of children with brainstem CMs.

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Editorial

Achondroplasia

Arnold H. Menezes