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Nikita G. Alexiades, Edward S. Ahn, Jeffrey P. Blount, Douglas L. Brockmeyer, Samuel R. Browd, Gerald A. Grant, Gregory G. Heuer, Todd C. Hankinson, Bermans J. Iskandar, Andrew Jea, Mark D. Krieger, Jeffrey R. Leonard, David D. Limbrick Jr., Cormac O. Maher, Mark R. Proctor, David I. Sandberg, John C. Wellons III, Belinda Shao, Neil A. Feldstein and Richard C. E. Anderson

OBJECTIVE

Complications after complex tethered spinal cord (cTSC) surgery include infections and cerebrospinal fluid (CSF) leaks. With little empirical evidence to guide management, there is variability in the interventions undertaken to limit complications. Expert-based best practices may improve the care of patients undergoing cTSC surgery. Here, authors conducted a study to identify consensus-driven best practices.

METHODS

The Delphi method was employed to identify consensual best practices. A literature review regarding cTSC surgery together with a survey of current practices was distributed to 17 board-certified pediatric neurosurgeons. Thirty statements were then formulated and distributed to the group. Results of the second survey were discussed during an in-person meeting leading to further consensus, which was defined as ≥ 80% agreement on a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree).

RESULTS

Seventeen consensus-driven best practices were identified, with all participants willing to incorporate them into their practice. There were four preoperative interventions: (1, 2) asymptomatic AND symptomatic patients should be referred to urology preoperatively, (3, 4) routine preoperative urine cultures are not necessary for asymptomatic AND symptomatic patients. There were nine intraoperative interventions: (5) patients should receive perioperative cefazolin or an equivalent alternative in the event of allergy, (6) chlorhexidine-based skin preparation is the preferred regimen, (7) saline irrigation should be used intermittently throughout the case, (8) antibiotic-containing irrigation should be used following dural closure, (9) a nonlocking running suture technique should be used for dural closure, (10) dural graft overlay should be used when unable to obtain primary dural closure, (11) an expansile dural graft should be incorporated in cases of lipomyelomeningocele in which primary dural closure does not permit free flow of CSF, (12) paraxial muscles should be closed as a layer separate from the fascia, (13) routine placement of postoperative drains is not necessary. There were three postoperative interventions: (14) postoperative antibiotics are an option and, if given, should be discontinued within 24 hours; (15) patients should remain flat for at least 24 hours postoperatively; (16) routine use of abdominal binders or other compressive devices postoperatively is not necessary. One intervention was prioritized for additional study: (17) further study of additional gram-negative perioperative coverage is needed.

CONCLUSIONS

A modified Delphi technique was used to develop consensus-driven best practices for decreasing wound complications after cTSC surgery. Further study is required to determine if implementation of these practices will lead to reduced complications. Discussion through the course of this study resulted in the initiation of a multicenter study of gram-negative surgical site infections in cTSC surgery.

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Jaims Lim, Alan R. Tang, Campbell Liles, Alexander A. Hysong, Andrew T. Hale, Christopher M. Bonfield, Robert P. Naftel, John C. Wellons III and Chevis N. Shannon

OBJECTIVE

Many studies have aimed to determine the most clinically effective surgical intervention for hydrocephalus. However, the costs associated with each treatment option are poorly understood. In this study, the authors conducted a cost-effectiveness analysis, calculating the incremental cost-effectiveness ratio (ICER) of ventriculoperitoneal shunting (VPS), endoscopic third ventriculostomy (ETV), and ETV with choroid plexus cauterization (ETV/CPC) in an effort to better understand the clinical effectiveness and costs associated with treating hydrocephalus.

METHODS

The study cohort includes patients under the age of 18 who were initially treated for hydrocephalus between January 2012 and January 2015 at the authors’ institution. Overall treatment costs were calculated using patient-level hospitalization costs and professional fees reimbursable to the hospital and directly related to the initial and follow-up (postoperative day 1 to 12 months) treatment of hydrocephalus. TreeAge Pro was used to conduct the cost-effectiveness analyses.

RESULTS

A total of 147 patients were identified. Based on the initial intervention for hydrocephalus, their cases were classified as follows: 113 VPS, 14 ETV, and 20 ETV/CPC. During the initial intervention, VPS patients required a longer length of stay at 5.6 days, compared to ETV/CPC (3.35 days) and ETV (2.36 days) patients. Failure rates for all treatment options ranged from 29% to 45%, leading to recurrent hydrocephalus and additional surgical intervention between postoperative day 1 and 12 months. Cost-effectiveness analyses found ETV to be less costly and more clinically effective, with an ICER of $94,797 compared to VPS ($130,839) and ETV/CPC ($126,394). However, when stratified by etiology, VPS was found to be more clinically effective and cost-effective in both the myelomeningocele and posthemorrhagic hydrocephalus patient groups with an incremental cost per clinical unit of effectiveness (success or failure of intervention) of $76,620 compared to ETV and ETV/CPC. However, when assessing cases categorized as “other etiologies,” ETV was found to be more cost-effective per clinical unit, with an ICER of $60,061 compared to ETV/CPC ($93,350) and VPS ($142,135).

CONCLUSIONS

This study is one of the first attempts at quantifying the patient-level hospitalization costs associated with surgical management of hydrocephalus in pediatric patients treated in the United States. The results indicate that the conversation regarding CSF diversion techniques must be patient-specific and consider etiology as well as any previous surgical intervention. Again, these findings are short-run observations, and a long-term follow-up study should be conducted to assess the cost of treating hydrocephalus over the lifetime of a patient.

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Christopher M. Bonfield, Rachel Pellegrino, Jillian Berkman, Robert P. Naftel, Chevis N. Shannon and John C. Wellons III

OBJECTIVE

Both the American Association of Neurological Surgeons/Congress of Neurological Surgeons Joint Section on Pediatric Neurological Surgery (AANS/CNS Pediatric Section) and the International Society for Pediatric Neurosurgery (ISPN) annual meetings provide a platform for pediatric neurosurgeons to present, discuss, and disseminate current academic research. An ultimate goal of these meetings is to publish presented results in peer-reviewed journals. The purpose of the present study was to investigate the publication rates of oral presentations from the 2009, 2010, and 2011 AANS/CNS Pediatric Section and ISPN annual meetings in peer-reviewed journals.

METHODS

All oral presentations from the 2009, 2010, and 2011 AANS/CNS Pediatric Section and ISPN annual meetings were reviewed. Abstracts were obtained from the AANS/CNS Pediatric Section and ISPN conference proceedings, which are available online. Author and title information were used to search PubMed to identify those abstracts that had progressed to publication in peer-reviewed journals. The title of the journal, year of the publication, and authors’ country of origin were also recorded.

RESULTS

Overall, 60.6% of the presented oral abstracts from the AANS/CNS Pediatric Section meetings progressed to publication in peer-reviewed journals, as compared with 40.6% of the ISPN presented abstracts (p = 0.0001). The journals in which the AANS/CNS Pediatric Section abstract-based publications most commonly appeared were Journal of Neurosurgery: Pediatrics (52%), Child’s Nervous System (11%), and Journal of Neurosurgery (8%). The ISPN abstracts most often appeared in the journals Child’s Nervous System (29%), Journal of Neurosurgery: Pediatrics (14%), and Neurosurgery (9%). Overall, more than 90% of the abstract-based articles were published within 4 years after presentation of the abstracts on which they were based.

CONCLUSIONS

Oral abstract presentations at two annual pediatric neurosurgery meetings have publication rates in peer-reviewed journal comparable to those for oral abstracts at other national and international neurosurgery meetings. The vast majority of abstract-based papers are published within 4 years of the meeting at which the abstract was presented; however, the AANS/CNS Pediatric Section abstracts are published at a significantly higher rate than ISPN abstracts, which could indicate the different meeting sizes, research goals, and resources of US authors compared with those of authors from other countries.

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Michael C. Dewan, Jaims Lim, Stephen R. Gannon, David Heaner, Matthew C. Davis, Brandy Vaughn, Joshua J. Chern, Brandon G. Rocque, Paul Klimo Jr., John C. Wellons III and Robert P. Naftel

OBJECTIVE

It has been suggested that the treatment of infant hydrocephalus results in different craniometric changes depending upon whether ventriculoperitoneal shunt (VPS) placement or endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC) is performed. Without an objective and quantitative description of expected changes to the infant cranium and ventricles following ETV/CPC, asserting successful treatment of hydrocephalus is difficult. By comparing infants successfully treated via ETV/CPC or VPS surgery, the authors of this study aimed to define the expected postoperative cranial and ventricular alterations at the time of clinical follow-up.

METHODS

Patients who underwent successful treatment of hydrocephalus at 4 institutions with either VPS placement or ETV/CPC were matched in a 3:1 ratio on the basis of age and etiology. Commonly used cranial parameters (including head circumference [HC], HC z-score, fontanelle status, and frontooccipital horn ratio [FOHR]) were compared pre- and postoperatively between treatment cohorts. First, baseline preoperative values were compared to ensure cohort equivalence. Next, postoperative metrics, including the relative change in metrics, were compared between treatment groups using multivariate linear regression.

RESULTS

Across 4 institutions, 18 ETV/CPC-treated and 54 VPS-treated infants with hydrocephalus were matched and compared at 6 months postoperatively. The most common etiologies of hydrocephalus were myelomeningocele (61%), followed by congenital communicating hydrocephalus (17%), aqueductal stenosis (11%), and intraventricular hemorrhage (6%). The mean age at the time of CSF diversion was similar between ETV/CPC- and VPS-treated patients (3.4 vs 2.9 months; p = 0.69), as were all preoperative cranial hydrocephalus metrics (p > 0.05). Postoperatively, the ventricle size FOHR decreased significantly more following VPS surgery (−0.15) than following ETV/CPC (−0.02) (p < 0.001), yielding a lower postoperative FOHR in the VPS arm (0.42 vs 0.51; p = 0.01). The HC percentile was greater in the ETV/CPC cohort than in the VPS-treated patients (76th vs 54th percentile; p = 0.046). A significant difference in the postoperative z-score was not observed. With both treatment modalities, a bulging fontanelle reliably normalized at last follow-up.

CONCLUSIONS

Clinical and radiographic parameters following successful treatment of hydrocephalus in infants differed between ETV/CPC and VPS treatment. At 6 months post-ETV/CPC, ventricle size remained unchanged, whereas VPS-treated ventricles decreased to a near-normal FOHR. The HC growth control between the procedures was similar, although the final HC percentile may be lower after VPS. The fontanelle remained a reliable indicator of success for both treatments. This study establishes expected cranial and ventricular parameters following ETV/CPC, which may be used to guide preoperative counseling and postoperative decision making.

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Michael C. Dewan, Abbas Rattani, Rania Mekary, Laurence J. Glancz, Ismaeel Yunusa, Ronnie E. Baticulon, Graham Fieggen, John C. Wellons III, Kee B. Park and Benjamin C. Warf

OBJECTIVE

Hydrocephalus is one of the most common brain disorders, yet a reliable assessment of the global burden of disease is lacking. The authors sought a reliable estimate of the prevalence and annual incidence of hydrocephalus worldwide.

METHODS

The authors performed a systematic literature review and meta-analysis to estimate the incidence of congenital hydrocephalus by WHO region and World Bank income level using the MEDLINE/PubMed and Cochrane Database of Systematic Reviews databases. A global estimate of pediatric hydrocephalus was obtained by adding acquired forms of childhood hydrocephalus to the baseline congenital figures using neural tube defect (NTD) registry data and known proportions of posthemorrhagic and postinfectious cases. Adult forms of hydrocephalus were also examined qualitatively.

RESULTS

Seventy-eight articles were included from the systematic review, representative of all WHO regions and each income level. The pooled incidence of congenital hydrocephalus was highest in Africa and Latin America (145 and 316 per 100,000 births, respectively) and lowest in the United States/Canada (68 per 100,000 births) (p for interaction < 0.1). The incidence was higher in low- and middle-income countries (123 per 100,000 births; 95% CI 98–152 births) than in high-income countries (79 per 100,000 births; 95% CI 68–90 births) (p for interaction < 0.01). While likely representing an underestimate, this model predicts that each year, nearly 400,000 new cases of pediatric hydrocephalus will develop worldwide. The greatest burden of disease falls on the African, Latin American, and Southeast Asian regions, accounting for three-quarters of the total volume of new cases. The high crude birth rate, greater proportion of patients with postinfectious etiology, and higher incidence of NTDs all contribute to a case volume in low- and middle-income countries that outweighs that in high-income countries by more than 20-fold. Global estimates of adult and other forms of acquired hydrocephalus are lacking.

CONCLUSIONS

For the first time in a global model, the annual incidence of pediatric hydrocephalus is estimated. Low- and middle-income countries incur the greatest burden of disease, particularly those within the African and Latin American regions. Reliable incidence and burden figures for adult forms of hydrocephalus are absent in the literature and warrant specific investigation. A global effort to address hydrocephalus in regions with the greatest demand is imperative to reduce disease incidence, morbidity, mortality, and disparities of access to treatment.

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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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Abhaya V. Kulkarni, Jay Riva-Cambrin, Curtis J. Rozzelle, Robert P. Naftel, Jessica S. Alvey, Ron W. Reeder, Richard Holubkov, Samuel R. Browd, D. Douglas Cochrane, David D. Limbrick Jr., Tamara D. Simon, Mandeep Tamber, John C. Wellons III, William E. Whitehead and John R. W. Kestle

OBJECTIVE

High-quality data comparing endoscopic third ventriculostomy (ETV) with choroid plexus cauterization (CPC) to shunt and ETV alone in North America are greatly lacking. To address this, the Hydrocephalus Clinical Research Network (HCRN) conducted a prospective study of ETV+CPC in infants. Here, these prospective data are presented and compared to prospectively collected data from a historical cohort of infants treated with shunt or ETV alone.

METHODS

From June 2014 to September 2015, infants (corrected age ≤ 24 months) requiring treatment for hydrocephalus with anatomy suitable for ETV+CPC were entered into a prospective study at 9 HCRN centers. The rate of procedural failure (i.e., the need for repeat hydrocephalus surgery, hydrocephalus-related death, or major postoperative neurological deficit) was determined. These data were compared with a cohort of similar infants who were treated with either a shunt (n = 969) or ETV alone (n = 74) by creating matched pairs on the basis of age and etiology. These data were obtained from the existing prospective HCRN Core Data Project. All patients were observed for at least 6 months.

RESULTS

A total of 118 infants underwent ETV+CPC (median corrected age 1.3 months; common etiologies including myelomeningocele [30.5%], intraventricular hemorrhage of prematurity [22.9%], and aqueductal stenosis [21.2%]). The 6-month success rate was 36%. The most common complications included seizures (5.1%) and CSF leak (3.4%). Important predictors of treatment success in the survival regression model included older age (p = 0.002), smaller preoperative ventricle size (p = 0.009), and greater degree of CPC (p = 0.02). The matching algorithm resulted in 112 matched pairs for ETV+CPC versus shunt alone and 34 matched pairs for ETV+CPC versus ETV alone. ETV+CPC was found to have significantly higher failure rate than shunt placement (p < 0.001). Although ETV+CPC had a similar failure rate compared with ETV alone (p = 0.73), the matched pairs included mostly infants with aqueductal stenosis and miscellaneous other etiologies but very few patients with intraventricular hemorrhage of prematurity.

CONCLUSIONS

Within a large and broad cohort of North American infants, our data show that overall ETV+CPC appears to have a higher failure rate than shunt alone. Although the ETV+CPC results were similar to ETV alone, this comparison was limited by the small sample size and skewed etiological distribution. Within the ETV+CPC group, greater extent of CPC was associated with treatment success, thereby suggesting that there are subgroups who might benefit from the addition of CPC. Further work will focus on identifying these subgroups.

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Silky Chotai, Bradley S. Guidry, Emily W. Chan, Katherine D. Sborov, Stephen Gannon, Chevis Shannon, Christopher M. Bonfield, John C. Wellons III and Robert P. Naftel

OBJECTIVE

Readmission and return to operating room after surgery are increasingly being used as a proxy for quality of care. Nearly 60% of these readmissions are unplanned, which translates into billions of dollars in health care costs. The authors set out to analyze the incidence of readmission at their center, to define causes of unplanned readmission, and to determine the preoperative and surgical variables associated with readmissions following pediatric neurosurgery.

METHODS

A total of 536 children who underwent operations for neurosurgical diagnoses between 2012 and 2015 and who were later readmitted were included in the final analysis. Unplanned readmissions were defined to have occurred as a result of complications within 90 days after index surgery. Patient records were retrospectively reviewed to determine the primary diagnosis, surgery indication, and cause of readmission and return to operating room. The cost for index hospitalization, readmission episode, and total cost were derived based on the charges obtained from administrative data. Bivariate and multivariable analyses were conducted.

RESULTS

Of 536 patients readmitted in total, 17.9% (n = 96) were readmitted within 90 days. Of the overall readmissions, 11.9% (n = 64) were readmitted within 30 days, and 5.97% (n = 32) were readmitted between 31 and 90 days. The median duration between discharge and readmission was 20 days (first quartile [Q1]: 9 days, third quartile [Q3]: 36 days). The most common reason for readmission was shunt related (8.2%, n = 44), followed by wound infection (4.7%, n = 25). In the risk-adjusted multivariable logistic regression model for total 90-day readmission, patients with the following characteristics: younger age (p = 0.001, OR 0.886, 95% CI 0.824–0.952); “other” (nonwhite, nonblack) race (p = 0.024, OR 5.49, 95% CI 1.246–24.2); and those born preterm (p = 0.032, OR 2.1, 95% CI 1.1–4.12) had higher odds of being readmitted within 90 days after discharge. The total median cost for patients undergoing surgery in this study cohort was $11,520 (Q1: $7103, Q3: $19,264). For the patients who were readmitted, the median cost for a readmission episode was $8981 (Q1: $5051, Q3: $18,713).

CONCLUSIONS

Unplanned 90-day readmissions in pediatric neurosurgery are primarily due to CSF-related complications. Patients with the following characteristics: young age at presentation; “other” race; and children born preterm have a higher likelihood of being readmitted within 90 days after surgery. The median cost was > $8000, which suggests that the readmission episode can be as expensive as the index hospitalization. Clearly, readmission reduction has the potential for significant cost savings in pediatric neurosurgery. Future efforts, such as targeted education related to complication signs, should be considered in the attempt to reduce unplanned events. Given the single-center, retrospective study design, the results of this study are primarily applicable to this population and cannot necessarily be generalized to other institutions without further study.

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Abbas Rattani, Michael C. Dewan, Vickie Hannig, Robert P. Naftel, John C. Wellons III and Lori C. Jordan

The authors present a case of monozygotic twins with hereditary hemorrhagic telangiectasia (HHT) who experienced cerebral arteriovenous malformation (AVM) hemorrhage at a very young age. The clinical variables influencing HHT-related AVM rupture are discussed, and questions surrounding the timing of screening and intervention are explored. This is only the second known case of monozygotic HHT twins published in the medical literature, and the youngest pair of first-degree relatives to experience AVM-related cerebral hemorrhage. Evidence guiding the screening and management of familial HHT is lacking, and cases such as this underscore the need for objective and validated protocols.