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Tamara D. Simon, Matthew P. Kronman, Kathryn B. Whitlock, Nancy E. Gove, Nicole Mayer-Hamblett, Samuel R. Browd, D. Douglas Cochrane, Richard Holubkov, Abhaya V. Kulkarni, Marcie Langley, David D. Limbrick Jr., Thomas G. Luerssen, W. Jerry Oakes, Jay Riva-Cambrin, Curtis Rozzelle, Chevis Shannon, Mandeep Tamber, John C. Wellons III, William E. Whitehead and John R. W. Kestle

OBJECTIVE

CSF shunt infection requires both surgical and antibiotic treatment. Surgical treatment includes either total shunt removal with external ventricular drain (EVD) placement followed by new shunt insertion, or distal shunt externalization followed by new shunt insertion once the CSF is sterile. Antibiotic treatment includes the administration of intravenous antibiotics. The Hydrocephalus Clinical Research Network (HCRN) registry provides a unique opportunity to understand reinfection following treatment for CSF shunt infection. This study examines the association of surgical and antibiotic decisions in the treatment of first CSF shunt infection with reinfection.

METHODS

A prospective cohort study of children undergoing treatment for first CSF infection at 7 HCRN hospitals from April 2008 to December 2012 was performed. The HCRN consensus definition was used to define CSF shunt infection and reinfection. The key surgical predictor variable was surgical approach to treatment for CSF shunt infection, and the key antibiotic treatment predictor variable was intravenous antibiotic selection and duration. Cox proportional hazards models were constructed to address the time-varying nature of the characteristics associated with shunt surgeries.

RESULTS

Of 233 children in the HCRN registry with an initial CSF shunt infection during the study period, 38 patients (16%) developed reinfection over a median time of 44 days (interquartile range [IQR] 19–437). The majority of initial CSF shunt infections were treated with total shunt removal and EVD placement (175 patients; 75%). The median time between infection surgeries was 15 days (IQR 10–22). For the subset of 172 infections diagnosed by CSF culture, the mean ± SD duration of antibiotic treatment was 18.7 ± 12.8 days. In all Cox proportional hazards models, neither surgical approach to infection treatment nor overall intravenous antibiotic duration was independently associated with reinfection. The only treatment decision independently associated with decreased infection risk was the use of rifampin. While this finding did not achieve statistical significance, in all 5 Cox proportional hazards models both surgical approach (other than total shunt removal at initial CSF shunt infection) and nonventriculoperitoneal shunt location were consistently associated with a higher hazard of reinfection, while the use of ultrasound was consistently associated with a lower hazard of reinfection.

CONCLUSIONS

Neither surgical approach to treatment nor antibiotic duration was associated with reinfection risk. While these findings did not achieve statistical significance, surgical approach other than total removal at initial CSF shunt infection was consistently associated with a higher hazard of reinfection in this study and suggests the feasibility of controlling and standardizing the surgical approach (shunt removal with EVD placement). Considerably more variation and equipoise exists in the duration and selection of intravenous antibiotic treatment. Further consideration should be given to the use of rifampin in the treatment of CSF shunt infection. High-quality studies of the optimal duration of antibiotic treatment are critical to the creation of evidence-based guidelines for CSF shunt infection treatment.

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John C. Wellons III, Chevis N. Shannon, Richard Holubkov, Jay Riva-Cambrin, Abhaya V. Kulkarni, David D. Limbrick Jr., William Whitehead, Samuel Browd, Curtis Rozzelle, Tamara D. Simon, Mandeep S. Tamber, W. Jerry Oakes, James Drake, Thomas G. Luerssen, John Kestle and For the Hydrocephalus Clinical Research Network

OBJECTIVE

Previous Hydrocephalus Clinical Research Network (HCRN) retrospective studies have shown a 15% difference in rates of conversion to permanent shunts with the use of ventriculosubgaleal shunts (VSGSs) versus ventricular reservoirs (VRs) as temporization procedures in the treatment of hydrocephalus due to high-grade intraventricular hemorrhage (IVH) of prematurity. Further research in the same study line revealed a strong influence of center-specific decision-making on shunt outcomes. The primary goal of this prospective study was to standardize decision-making across centers to determine true procedural superiority, if any, of VSGS versus VR as a temporization procedure in high-grade IVH of prematurity.

METHODS

The HCRN conducted a prospective cohort study across 6 centers with an approximate 1.5- to 3-year accrual period (depending on center) followed by 6 months of follow-up. Infants with premature birth, who weighed less than 1500 g, had Grade 3 or 4 IVH of prematurity, and had more than 72 hours of life expectancy were included in the study. Based on a priori consensus, decisions were standardized regarding the timing of initial surgical treatment, upfront shunt versus temporization procedure (VR or VSGS), and when to convert a VR or VSGS to a permanent shunt. Physical examination assessment and surgical technique were also standardized. The primary outcome was the proportion of infants who underwent conversion to a permanent shunt. The major secondary outcomes of interest included infection and other complication rates.

RESULTS

One hundred forty-five premature infants were enrolled and met criteria for analysis. Using the standardized decision rubrics, 28 infants never reached the threshold for treatment, 11 initially received permanent shunts, 4 were initially treated with endoscopic third ventriculostomy (ETV), and 102 underwent a temporization procedure (36 with VSGSs and 66 with VRs). The 2 temporization cohorts were similar in terms of sex, race, IVH grade, head (orbitofrontal) circumference, and ventricular size at temporization. There were statistically significant differences noted between groups in gestational age, birth weight, and bilaterality of clot burden that were controlled for in post hoc analysis. By Kaplan-Meier analysis, the 180-day rates of conversion to permanent shunts were 63.5% for VSGS and 74.0% for VR (p = 0.36, log-rank test). The infection rate for VSGS was 14% (5/36) and for VR was 17% (11/66; p = 0.71). The overall compliance rate with the standardized decision rubrics was noted to be 90% for all surgeons.

CONCLUSIONS

A standardized protocol was instituted across all centers of the HCRN. Compliance was high. Choice of temporization techniques in premature infants with IVH does not appear to influence rates of conversion to permanent ventricular CSF diversion. Once management decisions and surgical techniques are standardized across HCRN sites, thus minimizing center effect, the observed difference in conversion rates between VSGSs and VRs is mitigated.

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Brandon A. Sherrod, Anastasia A. Arynchyna, James M. Johnston, Curtis J. Rozzelle, Jeffrey P. Blount, W. Jerry Oakes and Brandon G. Rocque

OBJECTIVE

Surgical site infection (SSI) following CSF shunt operations has been well studied, yet risk factors for nonshunt pediatric neurosurgery are less well understood. The purpose of this study was to determine SSI rates and risk factors following nonshunt pediatric neurosurgery using a nationwide patient cohort and an institutional data set specifically for better understanding SSI.

METHODS

The authors reviewed the American College of Surgeons National Surgical Quality Improvement Program–Pediatric (ACS NSQIP-P) database for the years 2012–2014, including all neurosurgical procedures performed on pediatric patients except CSF shunts and hematoma evacuations. SSI included deep (intracranial abscesses, meningitis, osteomyelitis, and ventriculitis) and superficial wound infections. The authors performed univariate analyses of SSI association with procedure, demographic, comorbidity, operative, and hospital variables, with subsequent multivariate logistic regression analysis to determine independent risk factors for SSI within 30 days of the index procedure. A similar analysis was performed using a detailed institutional infection database from Children's of Alabama (COA).

RESULTS

A total of 9296 nonshunt procedures were identified in NSQIP-P with an overall 30-day SSI rate of 2.7%. The 30-day SSI rate in the COA institutional database was similar (3.3% of 1103 procedures, p = 0.325). Postoperative time to SSI in NSQIP-P and COA was 14.6 ± 6.8 days and 14.8 ± 7.3 days, respectively (mean ± SD). Myelomeningocele (4.3% in NSQIP-P, 6.3% in COA), spine (3.5%, 4.9%), and epilepsy (3.4%, 3.1%) procedure categories had the highest SSI rates by procedure category in both NSQIP-P and COA. Independent SSI risk factors in NSQIP-P included postoperative pneumonia (OR 4.761, 95% CI 1.269–17.857, p = 0.021), immune disease/immunosuppressant use (OR 3.671, 95% CI 1.371–9.827, p = 0.010), cerebral palsy (OR 2.835, 95% CI 1.463–5.494, p = 0.002), emergency operation (OR 1.843, 95% CI 1.011–3.360, p = 0.046), spine procedures (OR 1.673, 95% CI 1.036–2.702, p = 0.035), acquired CNS abnormality (OR 1.620, 95% CI 1.085–2.420, p = 0.018), and female sex (OR 1.475, 95% CI 1.062–2.049, p = 0.021). The only COA factor independently associated with SSI in the COA database included clean-contaminated wound classification (OR 3.887, 95% CI 1.354–11.153, p = 0.012), with public insurance (OR 1.966, 95% CI 0.957–4.041, p = 0.066) and spine procedures (OR 1.982, 95% CI 0.955–4.114, p = 0.066) approaching significance. Both NSQIP-P and COA multivariate model C-statistics were > 0.7.

CONCLUSIONS

The NSQIP-P SSI rates, but not risk factors, were similar to data from a single center.

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William E. Whitehead, Jay Riva-Cambrin, Abhaya V. Kulkarni, John C. Wellons III, Curtis J. Rozzelle, Mandeep S. Tamber, David D. Limbrick Jr., Samuel R. Browd, Robert P. Naftel, Chevis N. Shannon, Tamara D. Simon, Richard Holubkov, Anna Illner, D. Douglas Cochrane, James M. Drake, Thomas G. Luerssen, W. Jerry Oakes and John R. W. Kestle

OBJECTIVE

Accurate placement of ventricular catheters may result in prolonged shunt survival, but the best target for the hole-bearing segment of the catheter has not been rigorously defined. The goal of the study was to define a target within the ventricle with the lowest risk of shunt failure.

METHODS

Five catheter placement variables (ventricular catheter tip location, ventricular catheter tip environment, relationship to choroid plexus, catheter tip holes within ventricle, and crosses midline) were defined, assessed for interobserver agreement, and evaluated for their effect on shunt survival in univariate and multivariate analyses. De-identified subjects from the Shunt Design Trial, the Endoscopic Shunt Insertion Trial, and a Hydrocephalus Clinical Research Network study on ultrasound-guided catheter placement were combined (n = 858 subjects, all first-time shunt insertions, all patients < 18 years old). The first postoperative brain imaging study was used to determine ventricular catheter placement for each of the catheter placement variables.

RESULTS

Ventricular catheter tip location, environment, catheter tip holes within the ventricle, and crosses midline all achieved sufficient interobserver agreement (κ > 0.60). In the univariate survival analysis, however, only ventricular catheter tip location was useful in distinguishing a target within the ventricle with a survival advantage (frontal horn; log-rank, p = 0.0015). None of the other catheter placement variables yielded a significant survival advantage unless they were compared with catheter tips completely not in the ventricle. Cox regression analysis was performed, examining ventricular catheter tip location with age, etiology, surgeon, decade of surgery, and catheter entry site (anterior vs posterior). Only age (p < 0.001) and entry site (p = 0.005) were associated with shunt survival; ventricular catheter tip location was not (p = 0.37). Anterior entry site lowered the risk of shunt failure compared with posterior entry site by approximately one-third (HR 0.65, 95% CI 0.51–0.83).

CONCLUSIONS

This analysis failed to identify an ideal target within the ventricle for the ventricular catheter tip. Unexpectedly, the choice of an anterior versus posterior catheter entry site was more important in determining shunt survival than the location of the ventricular catheter tip within the ventricle. Entry site may represent a modifiable risk factor for shunt failure, but, due to inherent limitations in study design and previous clinical research on entry site, a randomized controlled trial is necessary before treatment recommendations can be made.

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Douglas L. Brockmeyer, W. Jerry Oakes, Curtis Rozzelle, James Johnston, Brandon G. Rocque, Richard C. E. Anderson, Neil Feldstein, Jonathan Martin, Gerald F. Tuite, Luis Rodriguez, Nicholas Wetjen, Phillip Aldana, Dave Pincus, Phillip Storm, Mark R. Proctor and Sean Lew

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William E. Whitehead, Jay Riva-Cambrin, John C. Wellons III, Abhaya V. Kulkarni, Samuel Browd, David Limbrick, Curtis Rozzelle, Mandeep S. Tamber, Tamara D. Simon, Chevis N. Shannon, Richard Holubkov, W. Jerry Oakes, Thomas G. Luerssen, Marion L. Walker, James M. Drake and John R. W. Kestle

Object

Shunt survival may improve when ventricular catheters are placed into the frontal horn or trigone of the lateral ventricle. However, techniques for accurate catheter placement have not been developed. The authors recently reported a prospective study designed to test the accuracy of catheter placement with the assistance of intraoperative ultrasound, but the results were poor (accurate placement in 59%). A major reason for the poor accurate placement rate was catheter movement that occurred between the time of the intraoperative ultrasound image and the first postoperative scan (33% of cases). The control group of non–ultrasound using surgeons also had a low rate of accurate placement (accurate placement in 49%). The authors conducted an exploratory post hoc analysis of patients in their ultrasound study to identify factors associated with either catheter movement or poor catheter placement so that improved surgical techniques for catheter insertion could be developed.

Methods

The authors investigated the following risk factors for catheter movement and poor catheter placement: age, ventricular size, cortical mantle thickness, surgeon experience, surgeon experience with ultrasound prior to trial, shunt entry site, shunt hardware at entry site, ventricular catheter length, and use of an ultrasound probe guide for catheter insertion. Univariate analysis followed by multivariate logistic regression models were used to determine which factors were independent risk factors for either catheter movement or inaccurate catheter location.

Results

In the univariate analyses, only age < 6 months was associated with catheter movement (p = 0.021); cortical mantle thickness < 1 cm was near-significant (p = 0.066). In a multivariate model, age remained significant after adjusting for cortical mantle thickness (OR 8.35, exact 95% CI 1.20–infinity). Univariate analyses of factors associated with inaccurate catheter placement showed that age < 6 months (p = 0.001) and a posterior shunt entry site (p = 0.021) were both associated with poor catheter placement. In a multivariate model, both age < 6 months and a posterior shunt entry site were independent risk factors for poor catheter placement (OR 4.54, 95% CI 1.80–11.42, and OR 2.59, 95% CI 1.14–5.89, respectively).

Conclusions

Catheter movement and inaccurate catheter placement are both more likely to occur in young patients (< 6 months). Inaccurate catheter placement is also more likely to occur in cases involving a posterior shunt entry site than those involving an anterior shunt entry site. Future clinical studies aimed at improving shunt placement techniques must consider the effects of young age and choice of entry site on catheter location.

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William E. Whitehead, Jay Riva-Cambrin, John C. Wellons III, Abhaya V. Kulkarni, Richard Holubkov, Anna Illner, W. Jerry Oakes, Thomas G. Luerssen, Marion L. Walker, James M. Drake and John R. W. Kestle

Object

Cerebrospinal fluid shunt ventricular catheters inserted into the frontal horn or trigone are associated with prolonged shunt survival. Developing surgical techniques for accurate catheter insertion could, therefore, be beneficial to patients. This study was conducted to determine if the rate of accurate catheter location with intraoperative ultrasound guidance could exceed 80%.

Methods

The authors conducted a prospective, multicenter study of children (< 18 years) requiring first-time treatment for hydrocephalus with a ventriculoperitoneal shunt. Using intraoperative ultrasound, surgeons were required to target the frontal horn or trigone for catheter tip placement. An intraoperative ultrasound image was obtained at the time of catheter insertion. Ventricular catheter location, the primary outcome measure, was determined from the first postoperative image. A control group of patients treated by nonultrasound surgeons (conventional surgeons) were enrolled using the same study criteria. Conventional shunt surgeons also agreed to target the frontal horn or trigone for all catheter insertions. Patients were triaged to participating surgeons based on call schedules at each center. A pediatric neuroradiologist blinded to method of insertion, center, and surgeon determined ventricular catheter tip location.

Results

Eleven surgeons enrolled as ultrasound surgeons and 6 as conventional surgeons. Between February 2009 and February 2010, 121 patients were enrolled at 4 Hydrocephalus Clinical Research Network centers. Experienced ultrasound surgeons (> 15 cases prior to study) operated on 67 patients; conventional surgeons operated on 52 patients. Experienced ultrasound surgeons achieved accurate catheter location in 39 (59%) of 66 patients, 95% CI (46%–71%). Intraoperative ultrasound images were compared with postoperative scans. In 32.7% of cases, the catheter tip moved from an accurate location on the intraoperative ultrasound image to an inaccurate location on the postoperative study. This was the most significant factor affecting accuracy. In comparison, conventional surgeons achieved accurate location in 24 (49.0%) of 49 cases (95% CI [34%–64%]). The shunt survival rate at 1 year was 70.8% in the experienced ultrasound group and 66.9% in the conventional group (p = 0.66). Ultrasound surgeons had more catheters surrounded by CSF (30.8% vs 6.1%, p = 0.0012) and away from the choroid plexus (72.3% vs 58.3%, p = 0.12), and fewer catheters in the brain (3% vs 22.4%, p = 0.0011) and crossing the midline (4.5% vs 34.7%, p < 0.001), but they had a higher proportion of postoperative pseudomeningocele (10.1% vs 3.8%, p = 0.30), wound dehiscence (5.8% vs 0%, p = 0.13), CSF leak (10.1% vs 1.9%, p = 0.14), and shunt infection (11.6% vs 5.8%, p = 0.35).

Conclusions

Ultrasound-guided shunt insertion as performed in this study was unable to consistently place catheters into the frontal horn or trigone. The technique is safe and achieves outcomes similar to other conventional shunt insertion techniques. Further efforts to improve accurate catheter location should focus on prevention of catheter migration that occurs between intraoperative placement and postoperative imaging. Clinical trial registration no.: NCT01007786 (ClinicalTrials.gov).

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Robert P. Naftel, R. Shane Tubbs, Joshua Y. Menendez, John C. Wellons III, Ian F. Pollack and W. Jerry Oakes

Object

The effects of posterior fossa decompression on Chiari malformation Type I–induced syringomyelia have been well described. However, treatment of worsening syringomyelia after Chiari decompression remains enigmatic. This paper defines patient and clinical characteristics as well as treatment and postoperative radiological and clinical outcomes in patients experiencing this complication.

Methods

The authors performed a retrospective review of patients at the Children's Hospital of Pittsburgh and Children's of Alabama who developed worsening syringomyelia after Chiari decompression was performed.

Results

Fourteen children (age range 8 months to 15 years), 7 of whom had preoperative syringomyelia, underwent posterior fossa decompression. Aseptic meningitis (n = 3) and bacterial meningitis (n = 2) complicated 5 cases (4 of these patients were originally treated at outside hospitals). Worsening syringomyelia presented a median of 1.4 years (range 0.2–10.3 years) after the primary decompression. Ten children presented with new, recurrent, or persistent symptoms, and 4 were asymptomatic. Secondary Chiari decompression was performed in 11 of the 14 children. The other 3 children were advised to undergo secondary decompression. A structural cause for each failed primary Chiari decompression (for example, extensive scarring, suture in the obex, arachnoid web, residual posterior arch of C-1, and no duraplasty) was identified at the secondary operation. After secondary decompression, 8 patients' symptoms completely resolved, 1 patient's condition stabilized, and 2 patients remained asymptomatic. Radiologically, 10 of the 11 children had a decrease in the size of their syringes, and 1 child experienced no change (but improved clinically). The median follow-up from initial Chiari decompression was 3.1 years (range 0.8–14.1 years) and from secondary decompression, 1.3 years (range 0.3–4.5 years). No patient underwent syringopleural shunting or other nonposterior fossa treatment for syringomyelia.

Conclusions

Based on the authors' experience, children with worsening syringomyelia after decompression for Chiari malformation Type I generally have a surgically remediable structural etiology, and secondary exploration and decompression should be considered.

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Robert P. Naftel, Nicole A. Safiano, Michael Falola, Jeffrey P. Blount, W. Jerry Oakes and John C. Wellons III

Object

Children experiencing frequent shunt failure consume medical resources and represent a disproportionate level of morbidity in hydrocephalus care. While biological causes of frequent shunt failure may exist, this study analyzed demographic and socioeconomic patient characteristics associated with frequent shunt failure.

Methods

A survey of 294 caregivers of children with shunt-treated hydrocephalus provided demographic and socioeconomic characteristics. Children experiencing at least 10 shunt failures were considered frequent shunt-failure patients. Multivariate regression models were used to control for variables.

Results

Frequent shunt failure was experienced by 9.5% of the patients (28 of 294). By univariate analysis, white race (p = 0.006), etiology of hydrocephalus (p = 0.022), years-with-shunt (p < 0.0001), and surgeon (p = 0.02) were associated with frequent shunt failure. Upon multivariate analysis, white race remained the key independent factor associated with frequent shunt failure (OR 5.8, 95% CI 1.2–27.8, p = 0.027). Race acted independently from socioeconomic factors, including income, level of education, and geographic location, and clinical factors, such as etiology of hydrocephalus, surgeon, and years-with-shunt. Additionally, after multivariate analysis surgeon and years-with-shunt remained associated with frequent shunt failure (p = 0.043 and p = 0.0098, respectively), although etiology of hydrocephalus was no longer associated (p = 0.1).

Conclusions

White race was the primary independent factor associated with frequent shunt failure. Because races use health care differently and the diagnosis of shunt failure is often subjective, a disparity in diagnosis and treatment has arisen. These findings call for objective criteria for the preoperative and intraoperative diagnosis of shunt failure.

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R. Shane Tubbs, Mitchel Muhleman, Samuel G. McClugage, Marios Loukas, Joseph H. Miller, Joshua J. Chern, Curtis J. Rozzelle, W. Jerry Oakes and Aaron A. Cohen-Gadol

Cysts of the choroidal fissure are often incidentally identified. Symptoms from such cysts appear to be exceedingly rare. Herein, the authors report a case series of symptomatic enlargement of choroidal fissure cysts that were surgically treated. Although cysts of the choroidal fissure do not normally become symptomatic, the neurosurgeon should be aware of such a complication. Based on the authors' experience, surgical fenestration of such cysts has good long-term results.