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Editorial. Spinning all around

John R. W. Kestle

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What to do with an incidental finding of a fused sagittal suture: a modified Delphi study

Sarah N. Chiang, Jocelyn Reckford, Allyson L. Alexander, Craig B. Birgfeld, Christopher M. Bonfield, Daniel E. Couture, Lisa R. David, Brooke French, Barbu Gociman, Jesse A. Goldstein, Michael S. Golinko, John R. W. Kestle, Amy Lee, Suresh N. Magge, Ian F. Pollack, S. Alex Rottgers, Christopher M. Runyan, Matthew D. Smyth, C. Corbett Wilkinson, Gary B. Skolnick, Jennifer M. Strahle, and Kamlesh B. Patel

OBJECTIVE

As many as 5% of normocephalic children may have a prematurely fused sagittal suture, yet the clinical significance and best course of management of this finding remain unclear. Providers in the Synostosis Research Group were surveyed to create a multicenter consensus on an optimal treatment and monitoring algorithm for this condition.

METHODS

A four-round modified Delphi method was utilized. The first two rounds consisted of anonymous surveys distributed to 10 neurosurgeons and 9 plastic surgeons with expertise in craniosynostosis across 9 institutions, and presented 3 patients (aged 3 years, 2 years, and 2 months) with incidentally discovered fused sagittal sutures, normal cephalic indices, and no parietal dysmorphology. Surgeons were queried about their preferred term for this entity and how best to manage these patients. Results were synthesized to create a treatment algorithm. The third and fourth feedback rounds consisted of open discussion of the algorithm until no further concerns arose.

RESULTS

Most surgeons preferred the term "premature fusion of the sagittal suture" (93%). At the conclusion of the final round, all surgeons agreed to not operate on the 3- and 2-year-old patients unless symptoms of intracranial hypertension or papilledema were present. In contrast, 50% preferred to operate on the 2-month-old. However, all agreed to utilize shared decision-making, taking into account any concerns about future head shape and neurodevelopment. Panelists agreed that patients over 18 months of age without signs or symptoms suggesting elevated intracranial pressure (ICP) should not undergo surgical treatment.

CONCLUSIONS

Through the Delphi method, a consensus regarding management of premature fusion of the sagittal suture was obtained from a panel of North American craniofacial surgeons. Without signs or symptoms of ICP elevation, surgery is not recommended in patients over 18 months of age. However, for children younger than 18 months, surgery should be discussed with caregivers using a shared decision-making process.

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A re-evaluation of the Endoscopic Third Ventriculostomy Success Score: a Hydrocephalus Clinical Research Network study

Leonard H. Verhey, Abhaya V. Kulkarni, Ron W. Reeder, Jay Riva-Cambrin, Hailey Jensen, Ian F. Pollack, Brandon G. Rocque, Mandeep S. Tamber, Patrick J. McDonald, Mark D. Krieger, Jonathan A. Pindrik, Jason S. Hauptman, Samuel R. Browd, William E. Whitehead, Eric M. Jackson, John C. Wellons III, Todd C. Hankinson, Jason Chu, David D. Limbrick Jr., Jennifer M. Strahle, John R. W. Kestle, and for the Hydrocephalus Clinical Research Network

OBJECTIVE

The Hydrocephalus Clinical Research Network (HCRN) conducted a prospective study 1) to determine if a new, better-performing version of the Endoscopic Third Ventriculostomy Success Score (ETVSS) could be developed, 2) to explore the performance characteristics of the original ETVSS in a modern endoscopic third ventriculostomy (ETV) cohort, and 3) to determine if the addition of radiological variables to the ETVSS improved its predictive abilities.

METHODS

From April 2008 to August 2019, children (corrected age ≤ 17.5 years) who underwent a first-time ETV for hydrocephalus were included in a prospective multicenter HCRN study. All children had at least 6 months of clinical follow-up and were followed since the index ETV in the HCRN Core Data Registry. Children who underwent choroid plexus cauterization were excluded. Outcome (ETV success) was defined as the lack of ETV failure within 6 months of the index procedure. Kaplan-Meier curves were constructed to evaluate time-dependent variables. Multivariable binary logistic models were built to evaluate predictors of ETV success. Model performance was evaluated with Hosmer-Lemeshow and Harrell’s C statistics.

RESULTS

Seven hundred sixty-one children underwent a first-time ETV. The rate of 6-month ETV success was 76%. The Hosmer-Lemeshow and Harrell’s C statistics of the logistic model containing more granular age and etiology categorizations did not differ significantly from a model containing the ETVSS categories. In children ≥ 12 months of age with ETVSSs of 50 or 60, the original ETVSS underestimated success, but this analysis was limited by a small sample size. Fronto-occipital horn ratio (p = 0.37), maximum width of the third ventricle (p = 0.39), and downward concavity of the floor of the third ventricle (p = 0.63) did not predict ETV success. A possible association between the degree of prepontine adhesions on preoperative MRI and ETV success was detected, but this did not reach statistical significance.

CONCLUSIONS

This modern, multicenter study of ETV success shows that the original ETVSS continues to demonstrate good predictive ability, which was not substantially improved with a new success score. There might be an association between preoperative prepontine adhesions and ETV success, and this needs to be evaluated in a future large prospective study.

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Shunt infection prevention practices in Hydrocephalus Clinical Research Network–Quality: a new quality improvement network for hydrocephalus management

Mandeep S. Tamber, Hailey Jensen, Jason Clawson, Nichol Nunn, John C. Wellons III, Jodi Smith, Jonathan E. Martin, and John R. W. Kestle

OBJECTIVE

Knowledge-based tools used to standardize perioperative care, such as the shunt infection prevention protocol of the Hydrocephalus Clinical Research Network (HCRN), have demonstrated their ability to reduce surgeon-based and center-based variations in outcomes and improve patient care. The mere presence of high-quality evidence, however, does not necessarily translate into improved patient outcomes owing to the implementation gap. To advance understanding of how knowledge-based tools are being utilized in the routine clinical care of children with hydrocephalus, the HCRN-Quality (HCRNq) network was started in 2019. With a focus on CSF shunt infection, the authors present baseline data regarding CSF shunt infection rates and current shunt infection prevention practices in use at HCRNq sites.

METHODS

Baseline shunt surgery practices, infection rate, and risk factor data were prospectively collected within HCRNq. No standard infection protocol was recommended, but site use of a protocol was implied if at least 3 of 6 common shunt infection prevention practices were used in > 80% of shunt surgical procedures. Univariable and multivariable analyses of shunt infection risk factors were performed.

RESULTS

Thirty sites accrued data on 2437 procedures between November 2019 and June 2021. The unadjusted infection rate across all sites was 3.9% (range 0%–13%) and did not differ among shunt insertion, shunt revision, or shunt insertion after infection. Protocol use was implied for only 15/30 centers and 60% of shunt operations. On univariable analysis, iodine/DuraPrep (OR 0.57, 95% CI 0.37–0.88, p = 0.02) and the use of an antibiotic-impregnated catheter in any segment of the shunt (or both) decreased infection risk (OR 0.53, 95% CI 0.34–0.82, p = 0.01). Iodine-based prep solutions (OR 0.56, 95% 0.36–0.86, p = 0.02) and the use of antibiotic-impregnated catheters (OR 0.52, 95% CI 0.34–0.81, p = 0.01) retained significance in the multivariable model, but no relationship between protocol use and infection risk was demonstrated in this baseline analysis.

CONCLUSIONS

The authors have demonstrated that children undergoing CSF shunt surgery at HCRNq sites share similar demographic characteristics with other large North American multicenter cohorts, with similar observed baseline infection rates and risk factors. Many centers have implemented standardized shunt infection prevention practices, but considerable practice variation remains. As such, there is an opportunity to decrease shunt infection rates in these centers through continued standardization of care.

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Comparison of outcomes in the management of abdominal pseudocyst in children with shunted hydrocephalus: a Hydrocephalus Clinical Research Network study

Vijay M. Ravindra, Hailey Jensen, Jay Riva-Cambrin, John C. Wellons III, David D. Limbrick Jr., Jonathan Pindrik, Eric M. Jackson, Ian F. Pollack, Todd C. Hankinson, Jason S. Hauptman, Mandeep S. Tamber, Abhaya V. Kulkarni, Brandon G. Rocque, Curtis Rozzelle, William E. Whitehead, Jason Chu, Mark D. Krieger, Tamara D. Simon, Ron Reeder, Patrick J. McDonald, Nichol Nunn, John R. W. Kestle, and for the Hydrocephalus Clinical Research Network

OBJECTIVE

Abdominal pseudocyst (APC) can cause distal site failure in children with ventriculoperitoneal shunts and is specifically designated as an infection in Hydrocephalus Clinical Research Network (HCRN) protocols. Specific management and outcomes of children with APCs have not been reported in a multicenter study. In this study, the authors investigated the management and outcomes of APC in children with shunted hydrocephalus who were treated at centers in the HCRN.

METHODS

The HCRN Registry was queried to identify children < 18 years old with shunts who were diagnosed with an APC (i.e., a loculated abdominal fluid collection containing the peritoneal catheter with abdominal distention and/or displacement of peritoneal contents). The primary outcome was shunt failure after APC treatment. The primary variable was reimplantation of the distal catheter after pseudocyst treatment back into the peritoneum versus implantation in a nonperitoneal site. Other risk factors for shunt failure after APC treatment and variability in APC management were investigated.

RESULTS

Among 141 children from 14 centers who underwent first-time management of an APC over a 14-year period, the median time from previous shunt surgery to APC diagnosis was 3.8 months. Overall, 17.7% of children had a positive culture: APC cultures were positive in 14.2% and CSF cultures in 15.6%. Six other children underwent shunt revision without removal; all underwent reoperation within 1 month. There was no difference in shunt survival (log-rank test, p = 0.42) or number of subsequent revisions within 6, 12, or 24 months for shunts reimplanted in the abdomen versus those implanted in a nonperitoneal location. Nonperitoneal implantation was associated with more noninfectious revisions (42.3% vs 22.9%, p = 0.019), whereas infection was more common after reimplantation in the abdomen (25.7% vs 7.0%, p = 0.003). Univariable analysis demonstrated that younger age at APC diagnosis (8.3 vs 12.2 years, p = 0.006) and prior shunt procedure within 12 weeks of APC diagnosis (59.5% vs 40.5%, p = 0.012) were associated with shunt failure after APC treatment. Multivariable modeling confirmed that prior shunt surgery within 12 weeks of APC diagnosis was independently associated with failure (HR 1.79 [95% CI 1.04–3.07], p = 0.035).

CONCLUSIONS

In the HCRN, APCs in the setting of CSF shunts are usually managed with externalization. Shunt surgery within 12 weeks of APC diagnosis was associated with risk of failure after APC treatment. Although no differences were found in overall shunt failure rate, noninfectious shunt revisions were more common in the nonperitoneal distal catheter sites, and infection was a more common reason for failure after reimplantation of the shunt in the abdomen.

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A multi-institutional survey on calvarial vault remodeling techniques for sagittal synostosis and outcomes analysis for patients treated at 12 months and older

David Chi, Ella Gibson, Sarah N. Chiang, Koeun Lee, Sybill D. Naidoo, Amy Lee, Craig Birgfeld, Ian F. Pollack, Jesse Goldstein, Michael Golinko, Christopher M. Bonfield, Faizi A. Siddiqi, John R. W. Kestle, Matthew D. Smyth, Kamlesh B. Patel, and the Synostosis Research Group (SynRG)

OBJECTIVE

Surgical treatment of sagittal craniosynostosis is challenging in older patients. This study aimed to assess the effect of increasing age on open surgical technique selection and patient outcomes using the multi-institutional Synostosis Research Group (SynRG) collaboration.

METHODS

Surgeons in SynRG were surveyed for key influences on their preferred open calvarial vault remodeling techniques at various patient ages: < 6, 6–12, and > 12 months. The SynRG database was then queried for open repairs of nonsyndromic sagittal craniosynostosis performed for patients older than 12 months of age. Perioperative measures, complications, and preoperative and postoperative cephalic indices were reviewed.

RESULTS

All surgeons preferred to treat patients at an earlier age, and most (89%) believed that less-optimal outcomes were achieved at ages older than 12 months. The modified pi procedure was the dominant technique in those younger than 12 months, while more involved open surgical techniques were performed for older patients, with a wide variety of open calvarial vault remodeling techniques used. Forty-four patients met inclusion criteria, with a mean (± SD) age at surgery of 29 ± 16 months. Eleven patients underwent parietal reshaping, 10 parietal-occipital switch, 9 clamshell craniotomy, 7 geometric parietal expansion, 6 modified pi procedure, and 1 parietal distraction. There were no readmissions, complications, or mortality within 30 days postoperatively. Patients’ cephalic indices improved a mean of 6.4% ± 4.0%, with a mean postoperative cephalic index of 74.2% ± 4.9%. Differences in postoperative cephalic index (p < 0.04) and hospital length of stay (p = 0.01) were significant between technique cohorts. Post hoc Tukey-Kramer analysis identified the parietal reshaping technique as being significantly associated with a reduced hospital length of stay.

CONCLUSIONS

Patient age is an important driver in technique selection, with surgeons selecting a more involved calvarial vault remodeling technique in older children. A variety of surgical techniques were analyzed, with the parietal reshaping technique being significantly associated with reduced length of stay; however, multiple perioperative factors may be contributory and require further analysis. When performed at high-volume centers by experienced pediatric neurosurgeons and craniofacial surgeons, open calvarial vault techniques can be a safe method for treating sagittal craniosynostosis in older children.

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Endoscopic third ventriculostomy in previously shunt-treated patients

Brandon G. Rocque, Hailey Jensen, Ron W. Reeder, Abhaya V. Kulkarni, Ian F. Pollack, John C. Wellons III, Robert P. Naftel, Eric M. Jackson, William E. Whitehead, Jonathan A. Pindrik, David D. Limbrick Jr., Patrick J. McDonald, Mandeep S. Tamber, Todd C. Hankinson, Jason S. Hauptman, Mark D. Krieger, Jason Chu, Tamara D. Simon, Jay Riva-Cambrin, John R. W. Kestle, Curtis J. Rozzelle, and for the Hydrocephalus Clinical Research Network

OBJECTIVE

Endoscopic third ventriculostomy (ETV) is an option for treatment of hydrocephalus, including for patients who have a history of previous treatment with CSF shunt insertion. The purpose of this study was to report the success of postshunt ETV by using data from a multicenter prospective registry.

METHODS

Prospectively collected data in the Hydrocephalus Clinical Research Network (HCRN) Core Data Project (i.e., HCRN Registry) were reviewed. Children who underwent ETV between 2008 and 2019 and had a history of previous treatment with a CSF shunt were included. A Kaplan-Meier survival curve was created for the primary outcome: time from postshunt ETV to subsequent CSF shunt placement or revision. Univariable Cox proportional hazards models were created to evaluate for an association between clinical and demographic variables and subsequent shunt surgery. Postshunt ETV complications were also identified and categorized.

RESULTS

A total of 203 children were included: 57% male and 43% female; 74% White, 23% Black, and 4% other race. The most common hydrocephalus etiologies were postintraventricular hemorrhage secondary to prematurity (56, 28%) and aqueductal stenosis (42, 21%). The ETV Success Score ranged from 10 to 80. The median patient age was 4.1 years. The overall success of postshunt ETV at 6 months was 41%. Only the surgeon’s report of a clear view of the basilar artery was associated with a lower likelihood of postshunt ETV failure (HR 0.43, 95% CI 0.23–0.82, p = 0.009). None of the following variables were associated with postshunt ETV success: age at the time of postshunt ETV, etiology of hydrocephalus, sex, race, ventricle size, number of previous shunt operations, ETV performed at time of shunt infection, and use of external ventricular drainage. Overall, complications were reported in 22% of patients, with CSF leak (8.6%) being the most common complication.

CONCLUSIONS

Postshunt ETV was successful in treating hydrocephalus, without subsequent need for a CSF shunt, in 41% of patients, with a clear view of the basilar artery being the only variable significantly associated with success. Complications occurred in 22% of patients. ETV is an option for treatment of hydrocephalus in children who have previously undergone shunt placement, but with a lower than expected likelihood of success.

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Hydrocephalus surveillance following CSF diversion: a modified Delphi study

David S. Hersh, Jonathan E. Martin, Ruth E. Bristol, Samuel R. Browd, Gerald Grant, Nalin Gupta, Todd C. Hankinson, Eric M. Jackson, John R. W. Kestle, Mark D. Krieger, Abhaya V. Kulkarni, Casey J. Madura, Jonathan Pindrik, Ian F. Pollack, Jeffrey S. Raskin, Jay Riva-Cambrin, Curtis J. Rozzelle, Jodi L. Smith, and John C. Wellons III

OBJECTIVE

Long-term follow-up is often recommended for patients with hydrocephalus, but the frequency of clinical follow-up, timing and modality of imaging, and duration of surveillance have not been clearly defined. Here, the authors used the modified Delphi method to identify areas of consensus regarding the modality, frequency, and duration of hydrocephalus surveillance following surgical treatment.

METHODS

Pediatric neurosurgeons serving as institutional liaisons to the Hydrocephalus Clinical Research Network (HCRN), or its implementation/quality improvement arm (HCRNq), were invited to participate in this modified Delphi study. Thirty-seven consensus statements were generated and distributed via an anonymous electronic survey, with responses structured as a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree). A subsequent, virtual meeting offered the opportunity for open discussion and modification of the statements in an effort to reach consensus (defined as ≥ 80% agreement or disagreement).

RESULTS

Nineteen pediatric neurosurgeons participated in the first round, after which 15 statements reached consensus. During the second round, 14 participants met virtually for review and discussion. Some statements were modified and 2 statements were combined, resulting in a total of 36 statements. At the conclusion of the session, consensus was achieved for 17 statements regarding the following: 1) the role of standardization; 2) preferred imaging modalities; 3) postoperative follow-up after shunt surgery (subdivided into immediate postoperative imaging, delayed postoperative imaging, routine clinical surveillance, and routine radiological surveillance); and 4) postoperative follow-up after an endoscopic third ventriculostomy. Consensus could not be achieved for 19 statements.

CONCLUSIONS

Using the modified Delphi method, 17 consensus statements were developed with respect to both clinical and radiological follow-up after a shunt or endoscopic third ventriculostomy. The frequency, modality, and duration of surveillance were addressed, highlighting areas in which no clear data exist to guide clinical practice. Although further studies are needed to evaluate the clinical utility and cost-effectiveness of hydrocephalus surveillance, the current study provides a framework to guide future efforts to develop standardized clinical protocols for the postoperative surveillance of patients with hydrocephalus. Ultimately, the standardization of hydrocephalus surveillance has the potential to improve patient care as well as optimize the use of healthcare resources.

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Endoscopic third ventriculostomy revision after failure of initial endoscopic third ventriculostomy and choroid plexus cauterization

Anastasia Arynchyna-Smith, Curtis J. Rozzelle, Hailey Jensen, Ron W. Reeder, Abhaya V. Kulkarni, Ian F. Pollack, John C. Wellons III, Robert P. Naftel, Eric M. Jackson, William E. Whitehead, Jonathan A. Pindrik, David D. Limbrick Jr., Patrick J. McDonald, Mandeep S. Tamber, Brent R. O’Neill, Jason S. Hauptman, Mark D. Krieger, Jason Chu, Tamara D. Simon, Jay Riva-Cambrin, John R. W. Kestle, Brandon G. Rocque, and for the Hydrocephalus Clinical Research Network

OBJECTIVE

Primary treatment of hydrocephalus with endoscopic third ventriculostomy (ETV) and choroid plexus cauterization (CPC) is well described in the neurosurgical literature, with wide reported ranges of success and complication rates. The purpose of this study was to describe the safety and efficacy of ETV revision after initial ETV+CPC failure.

METHODS

Prospectively collected data in the Hydrocephalus Clinical Research Network Core Data Project registry were reviewed. Children who underwent ETV+CPC as the initial treatment for hydrocephalus between 2013 and 2019 and in whom the initial ETV+CPC was completed (i.e., not abandoned) were included. Log-rank survival analysis (the primary analysis) was used to compare time to failure (defined as any other surgical treatment for hydrocephalus or death related to hydrocephalus) of initial ETV+CPC versus that of ETV revision by using random-effects modeling to account for the inclusion of patients in both the initial and revision groups. Secondary analysis compared ETV revision to shunt placement after failure of initial ETV+CPC by using the log-rank test, as well as shunt failure after ETV+CPC to that after ETV revision. Cox regression analysis was used to identify predictors of failure among children treated with ETV revision.

RESULTS

The authors identified 521 ETV+CPC procedures that met their inclusion criteria. Ninety-one children underwent ETV revision after ETV+CPC failure. ETV revision had a lower 1-year success rate than initial ETV+CPC (29.5% vs 45%, p < 0.001). ETV revision after initial ETV+CPC failure had a lower success rate than shunting (29.5% vs 77.8%, p < 0.001). Shunt survival after initial ETV+CPC failure was not significantly different from shunt survival after ETV revision failure (p = 0.963). Complication rates were similar for all examined surgical procedures (initial ETV+CPC, ETV revision, ventriculoperitoneal shunt [VPS] placement after ETV+CPC, and VPS placement after ETV revision). Only young age was predictive of ETV revision failure (p = 0.02).

CONCLUSIONS

ETV revision had a significantly lower 1-year success rate than initial ETV+CPC and VPS placement after ETV+CPC. Complication rates were similar for all studied procedures. Younger age, but not time since initial ETV+CPC, was a risk factor for ETV revision failure.

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The Hydrocephalus Clinical Research Network quality improvement initiative: the role of antibiotic-impregnated catheters and vancomycin wound irrigation

Jason Chu, Hailey Jensen, Richard Holubkov, Mark D. Krieger, Abhaya V. Kulkarni, Jay Riva-Cambrin, Curtis J. Rozzelle, David D. Limbrick Jr., John C. Wellons III, Samuel R. Browd, William E. Whitehead, Ian F. Pollack, Tamara D. Simon, Mandeep S. Tamber, Jason S. Hauptman, Jonathan Pindrik, Robert P. Naftel, Patrick J. McDonald, Todd C. Hankinson, Eric M. Jackson, Brandon G. Rocque, Ron Reeder, James M. Drake, John R. W. Kestle, and for the Hydrocephalus Clinical Research Network

OBJECTIVE

Two previous Hydrocephalus Clinical Research Network (HCRN) studies have demonstrated that compliance with a standardized CSF shunt infection protocol reduces shunt infections. In this third iteration, a simplified protocol consisting of 5 steps was implemented. This analysis provides an updated evaluation of protocol compliance and evaluates modifiable shunt infection risk factors.

METHODS

The new simplified protocol was implemented at HCRN centers on November 1, 2016, for all shunt procedures, excluding external ventricular drains, ventricular reservoirs, and subgaleal shunts. Procedures performed through December 31, 2019, were included (38 months). Compliance with the protocol, use of antibiotic-impregnated catheters (AICs), and other variables of interest were collected at the index operation. Outcome events for a minimum of 6 months postoperatively were recorded. The definition of infection was unchanged from the authors’ previous report.

RESULTS

A total of 4913 procedures were performed at 13 HCRN centers. The overall infection rate was 5.1%. Surgeons were compliant with all 5 steps of the protocol in 79.4% of procedures. The infection rate for the protocol alone was 8.1% and dropped to 4.9% when AICs were added. Multivariate analysis identified having ≥ 2 complex chronic conditions (odds ratio [OR] 1.76, 95% confidence interval [CI] 1.26–2.44, p = 0.01) and a history of prior shunt surgery within 12 weeks (OR 1.84, 95% CI 1.37–2.47, p < 0.01) as independent risk factors for shunt infection. The use of AICs (OR 0.70, 95% CI 0.50–0.97, p = 0.05) and vancomycin irrigation (OR 0.36, 95% CI 0.21–0.62, p < 0.01) were identified as independent factors protective against shunt infection.

CONCLUSIONS

The authors report the third iteration of their quality improvement protocol to reduce the risk of shunt infection. Compliance with the protocol was high. These updated data suggest that the incorporation of AICs is an important, modifiable infection prevention measure. Vancomycin irrigation was also identified as a protective factor but requires further study to better understand its role in preventing shunt infection.