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Andrew T. Hale, P. David Adelson, Gregory W. Albert, Philipp R. Aldana, Tord D. Alden, Richard C. E. Anderson, David F. Bauer, Christopher M. Bonfield, Douglas L. Brockmeyer, Joshua J. Chern, Daniel E. Couture, David J. Daniels, Susan R. Durham, Richard G. Ellenbogen, Ramin Eskandari, Timothy M. George, Gerald A. Grant, Patrick C. Graupman, Stephanie Greene, Jeffrey P. Greenfield, Naina L. Gross, Daniel J. Guillaume, Gregory G. Heuer, Mark Iantosca, Bermans J. Iskandar, Eric M. Jackson, James M. Johnston, Robert F. Keating, Jeffrey R. Leonard, Cormac O. Maher, Francesco T. Mangano, J. Gordon McComb, Thanda Meehan, Arnold H. Menezes, Brent O’Neill, Greg Olavarria, Tae Sung Park, John Ragheb, Nathan R. Selden, Manish N. Shah, Matthew D. Smyth, Scellig S. D. Stone, Jennifer M. Strahle, Scott D. Wait, John C. Wellons, William E. Whitehead, Chevis N. Shannon, David D. Limbrick Jr. and for the Park-Reeves Syringomyelia Research Consortium Investigators

OBJECTIVE

Factors associated with syrinx size in pediatric patients undergoing posterior fossa decompression (PFD) or PFD with duraplasty (PFDD) for Chiari malformation type I (CM-I) with syringomyelia (SM; CM-I+SM) are not well established.

METHODS

Using the Park-Reeves Syringomyelia Research Consortium registry, the authors analyzed variables associated with syrinx radiological outcomes in patients (< 20 years old at the time of surgery) with CM-I+SM undergoing PFD or PFDD. Syrinx resolution was defined as an anteroposterior (AP) diameter of ≤ 2 mm or ≤ 3 mm or a reduction in AP diameter of ≥ 50%. Syrinx regression or progression was defined using 1) change in syrinx AP diameter (≥ 1 mm), or 2) change in syrinx length (craniocaudal, ≥ 1 vertebral level). Syrinx stability was defined as a < 1-mm change in syrinx AP diameter and no change in syrinx length.

RESULTS

The authors identified 380 patients with CM-I+SM who underwent PFD or PFDD. Cox proportional hazards modeling revealed younger age at surgery and PFDD as being independently associated with syrinx resolution, defined as a ≤ 2-mm or ≤ 3-mm AP diameter or ≥ 50% reduction in AP diameter. Radiological syrinx resolution was associated with improvement in headache (p < 0.005) and neck pain (p < 0.011) after PFD or PFDD. Next, PFDD (p = 0.005), scoliosis (p = 0.007), and syrinx location across multiple spinal segments (p = 0.001) were associated with syrinx diameter regression, whereas increased preoperative frontal-occipital horn ratio (FOHR; p = 0.007) and syrinx location spanning multiple spinal segments (p = 0.04) were associated with syrinx length regression. Scoliosis (HR 0.38 [95% CI 0.16–0.91], p = 0.03) and smaller syrinx diameter (5.82 ± 3.38 vs 7.86 ± 3.05 mm; HR 0.60 [95% CI 0.34–1.03], p = 0.002) were associated with syrinx diameter stability, whereas shorter preoperative syrinx length (5.75 ± 4.01 vs 9.65 ± 4.31 levels; HR 0.21 [95% CI 0.12–0.38], p = 0.0001) and smaller pB-C2 distance (6.86 ± 1.27 vs 7.18 ± 1.38 mm; HR 1.44 [95% CI 1.02–2.05], p = 0.04) were associated with syrinx length stability. Finally, younger age at surgery (8.19 ± 5.02 vs 10.29 ± 4.25 years; HR 1.89 [95% CI 1.31–3.04], p = 0.01) was associated with syrinx diameter progression, whereas increased postoperative syrinx diameter (6.73 ± 3.64 vs 3.97 ± 3.07 mm; HR 3.10 [95% CI 1.67–5.76], p = 0.003), was associated with syrinx length progression. PFD versus PFDD was not associated with syrinx progression or reoperation rate.

CONCLUSIONS

These data suggest that PFDD and age are independently associated with radiological syrinx improvement, although forthcoming results from the PFDD versus PFD randomized controlled trial (NCT02669836, clinicaltrials.gov) will best answer this question.

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Jeremy S. Wetzel, Alex D. Waldman, Pavlos Texakalidis, Bryan Buster, Sheila R. Eshraghi, Jennifer Wheelus, Andrew Reisner and Joshua J. Chern

OBJECTIVE

The malfunction rates of and trends in various cerebrospinal fluid (CSF) shunt designs have been widely studied, but one area that has received little attention is the comparison of the peritoneal distal slit valve (DSV) shunt to other conventional valve (CV) type shunts. The literature that does exist comes from older case series that provide only indirect comparisons, and the conclusions are mixed. Here, the authors provide a direct comparison of the overall survival and failure trends of DSV shunts to those of other valve type shunts.

METHODS

Three hundred seventy-two new CSF shunts were placed in pediatric patients at the authors’ institution between January 2011 and December 2015. Only ventriculoperitoneal (VP) shunts were eligible for study inclusion. Ventriculoatrial, lumboperitoneal, cystoperitoneal, subdural-peritoneal, and spinal shunts were all excluded. Rates and patterns of shunt malfunction were compared, and survival curves were generated. Patterns of failure were categorized as proximal failure, distal failure, simultaneous proximal and distal (proximal+distal) failure, removal for infection, externalization for abdominal pseudocyst, and addition of a ventricular catheter for loculated hydrocephalus.

RESULTS

A total of 232 VP shunts were included in the final analysis, 115 DSV shunts and 117 CV shunts. There was no difference in the overall failure rate or time to failure between the two groups, and the follow-up period was statistically similar between the groups. The DSV group had a failure rate of 54% and a mean time to failure of 17.8 months. The CV group had a failure rate of 50% (p = 0.50) and a mean time to failure of 18.5 months (p = 0.56). The overall shunt survival curves for these two groups were similar; however, the location of failure was significantly different between the two groups. Shunts with DSVs had proportionately more distal failures than the CV group (34% vs 14%, respectively, p = 0.009). DSV shunts were also found to have proximal+distal catheter occlusions more frequently than CV shunts (23% vs 5%, respectively, p = 0.005). CV shunts were found to have significantly more proximal failures than the DSV shunts (53% vs 27%, p = 0.028). However, the only failure type that carried a statistically significant adjusted hazard ratio in a multivariate analysis was proximal+distal catheter obstruction (CV vs DSV shunt: HR 0.21, 95% CI 0.05–0.81).

CONCLUSIONS

There appears to be a difference in the location of catheter obstruction leading to the malfunction of shunts with DSVs compared to shunts with CVs; however, overall shunt survival is similar between the two. These failure types are also affected by other factors such etiology of hydrocephalus and endoscope use. The implications of these findings are unclear, and this topic warrants further investigation.

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Andrew Erwood, Rima S. Rindler, Mustafa Motiwala, Sonia Ajmera, Brandy Vaughn, Paul Klimo Jr. and Joshua J. Chern

OBJECTIVE

There are many known complications associated with CSF shunts. One of the more rare ones is a sterile abdominal pseudocyst due to decreased peritoneal absorption. This study was undertaken to detail the presentation, evaluation, and management of this unusual shunt-related event.

METHODS

Patients presenting with ventriculoperitoneal shunt (VPS)–related sterile abdominal pseudocysts treated at two institutions between 2013 and 2018 were included. Patients who had undergone abdominal surgery or shunt revisions within a 12-month period preceding presentation were excluded. Information was collected regarding clinical characteristics; hospital course, including surgical intervention(s); and any subsequent complications. Special attention was given to the eventual surgery after pseudocyst resolution, including the use of laparoscopy for peritoneal catheter placement, distal shunt conversion (i.e., in the atrium or pleural cavity), endoscopic third ventriculostomy, or shunt removal. The timing and nature of any subsequent shunt failures were also noted.

RESULTS

Twenty-eight patients met the study criteria, with a mean age of 10 years. The most common etiology of hydrocephalus was intraventricular hemorrhage of prematurity. All shunts were externalized at presentation. One shunt was removed without subsequent internalization. Distal catheters were re-internalized back into the peritoneal cavity in 11 patients (laparoscopy was used in 8 cases). Fourteen shunts were converted to a ventriculoatrial shunt (VAS), and two to a ventriculopleural (VPlS). Two VPSs failed due to a recurrent pseudocyst. The total all-cause failure rates at 1 year were as follows: 18% for VPSs and 50% for VASs.

CONCLUSIONS

Following treatment of a VPS-related sterile abdominal pseudocyst, laparoscopy-assisted placement of the distal catheter in the peritoneum is a viable and safe option for select patients, compared to a VAS or VPlS.

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Andrew B. Boucher and Joshua J. Chern

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Jennifer M. Strahle, Rukayat Taiwo, Christine Averill, James Torner, Chevis N. Shannon, Christopher M. Bonfield, Gerald F. Tuite, Tammy Bethel-Anderson, Jerrel Rutlin, Douglas L. Brockmeyer, John C. Wellons III, Jeffrey R. Leonard, Francesco T. Mangano, James M. Johnston, Manish N. Shah, Bermans J. Iskandar, Elizabeth C. Tyler-Kabara, David J. Daniels, Eric M. Jackson, Gerald A. Grant, Daniel E. Couture, P. David Adelson, Tord D. Alden, Philipp R. Aldana, Richard C. E. Anderson, Nathan R. Selden, Lissa C. Baird, Karin Bierbrauer, Joshua J. Chern, William E. Whitehead, Richard G. Ellenbogen, Herbert E. Fuchs, Daniel J. Guillaume, Todd C. Hankinson, Mark R. Iantosca, W. Jerry Oakes, Robert F. Keating, Nickalus R. Khan, Michael S. Muhlbauer, J. Gordon McComb, Arnold H. Menezes, John Ragheb, Jodi L. Smith, Cormac O. Maher, Stephanie Greene, Michael Kelly, Brent R. O’Neill, Mark D. Krieger, Mandeep Tamber, Susan R. Durham, Greg Olavarria, Scellig S. D. Stone, Bruce A. Kaufman, Gregory G. Heuer, David F. Bauer, Gregory Albert, Jeffrey P. Greenfield, Scott D. Wait, Mark D. Van Poppel, Ramin Eskandari, Timothy Mapstone, Joshua S. Shimony, Ralph G. Dacey Jr., Matthew D. Smyth, Tae Sung Park and David D. Limbrick Jr.

OBJECTIVE

Scoliosis is frequently a presenting sign of Chiari malformation type I (CM-I) with syrinx. The authors’ goal was to define scoliosis in this population and describe how radiological characteristics of CM-I and syrinx relate to the presence and severity of scoliosis.

METHODS

A large multicenter retrospective and prospective registry of pediatric patients with CM-I (tonsils ≥ 5 mm below the foramen magnum) and syrinx (≥ 3 mm in axial width) was reviewed for clinical and radiological characteristics of CM-I, syrinx, and scoliosis (coronal curve ≥ 10°).

RESULTS

Based on available imaging of patients with CM-I and syrinx, 260 of 825 patients (31%) had a clear diagnosis of scoliosis based on radiographs or coronal MRI. Forty-nine patients (5.9%) did not have scoliosis, and in 516 (63%) patients, a clear determination of the presence or absence of scoliosis could not be made. Comparison of patients with and those without a definite scoliosis diagnosis indicated that scoliosis was associated with wider syrinxes (8.7 vs 6.3 mm, OR 1.25, p < 0.001), longer syrinxes (10.3 vs 6.2 levels, OR 1.18, p < 0.001), syrinxes with their rostral extent located in the cervical spine (94% vs 80%, OR 3.91, p = 0.001), and holocord syrinxes (50% vs 16%, OR 5.61, p < 0.001). Multivariable regression analysis revealed syrinx length and the presence of holocord syrinx to be independent predictors of scoliosis in this patient cohort. Scoliosis was not associated with sex, age at CM-I diagnosis, tonsil position, pB–C2 distance (measured perpendicular distance from the ventral dura to a line drawn from the basion to the posterior-inferior aspect of C2), clivoaxial angle, or frontal-occipital horn ratio. Average curve magnitude was 29.9°, and 37.7% of patients had a left thoracic curve. Older age at CM-I or syrinx diagnosis (p < 0.0001) was associated with greater curve magnitude whereas there was no association between syrinx dimensions and curve magnitude.

CONCLUSIONS

Syrinx characteristics, but not tonsil position, were related to the presence of scoliosis in patients with CM-I, and there was an independent association of syrinx length and holocord syrinx with scoliosis. Further study is needed to evaluate the nature of the relationship between syrinx and scoliosis in patients with CM-I.

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Jeremy S. Wetzel, David P. Heaner, Brandon C. Gabel, R. Shane Tubbs and Joshua J. Chern

OBJECTIVE

The majority of children with myelomeningocele undergo implantation of CSF shunts. The efficacy of adding surveillance imaging to clinical evaluation during routine follow-up as a means to minimize the hazard associated with future shunt failure has not been thoroughly studied.

METHODS

A total of 300 spina bifida clinic visits during the calendar years between 2012 and 2016 were selected for this study (defined as the index clinic visit). Each index visit was preceded by a 6-month period during which no shunt evaluation of any kind was performed. At the index clinic visit, all patients were evaluated by a neurosurgeon. Seventy-four patients underwent previously scheduled surveillance CT or shunt series scans in addition to clinical evaluation (surveillance imaging group), and 226 patients did not undergo surveillance imaging (clinical evaluation group). Subsequent unexpected events, defined as emergency department visits, caregiver-requested clinic visits, and shunt revision surgeries were reviewed. The timing and likelihood of an unexpected event in each of the 2 groups were compared using Cox proportional hazard survival analysis. The rate of shunt revision surgery in the follow-up period as well as the associated outcomes and rate of complications were analyzed.

RESULTS

The clinical characteristics of the 2 groups were similar. In the clinical evaluation group, 4 of 226 (1.8%) patients underwent shunt revision based on clinical findings during the index visit, compared to 8 of 74 (10.8%) patients in the surveillance imaging group who underwent shunt revision based on clinical and imaging findings at that visit (p < 0.05). In the subsequent follow-up period, there were 74 unexpected events resulting in 10 shunt revisions in the clinical evaluation group, for an event rate of 33% and operation rate of 13.5%. In the surveillance imaging group there were 23 unexpected events resulting in 2 shunt revisions, for an event rate of 34.8% and an operation rate of 8.7%; neither difference was statistically significant. The complication rate for shunt revision surgery was also not different between the groups.

CONCLUSIONS

Obtaining predecided, routine surveillance imaging in children with myelomeningocele and shunted hydrocephalus resulted in more shunt revisions in asymptomatic patients. For patients who had negative results on surveillance imaging, the rate of shunt revision in the follow-up period was not significantly decreased compared to patients who underwent clinical examination only at the index visit.

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Andrew Reisner, Joshua J. Chern, Karen Walson, Natalie Tillman, Toni Petrillo-Albarano, Eric A. Sribnick, Laura S. Blackwell, Zaev D. Suskin, Chia-Yi Kuan and Atul Vats

OBJECTIVE

Evidence shows mixed efficacy of applying guidelines for the treatment of traumatic brain injury (TBI) in children. A multidisciplinary team at a children’s health system standardized intensive care unit–based TBI care using guidelines and best practices. The authors sought to investigate the impact of guideline implementation on outcomes.

METHODS

A multidisciplinary group developed a TBI care protocol based on published TBI treatment guidelines and consensus, which was implemented in March 2011. The authors retrospectively compared preimplementation outcomes (May 2009 to March 2011) and postimplementation outcomes (April 2011 to March 2014) among patients < 18 years of age admitted with severe TBI (Glasgow Coma Scale score ≤ 8) and potential survivability who underwent intracranial pressure (ICP) monitoring. Measures included mortality, hospital length of stay (LOS), ventilator LOS, critical ICP elevation time (percentage or total time that ICP was > 40 mm Hg), and survivor functionality at discharge (measured by the WeeFIM score). Data were analyzed using Student t-tests.

RESULTS

A total of 71 and 121 patients were included pre- and postimplementation, respectively. Mortality (32% vs 19%; p < 0.001) and length of critical ICP elevation (> 20 mm Hg; 26.3% vs 15%; p = 0.001) decreased after protocol implementation. WeeFIM discharge scores were not statistically different (57.6 vs 58.9; p = 0.9). Hospital LOS (median 19.6 days; p = 0.68) and ventilator LOS (median 10 days; p = 0.24) were unchanged.

CONCLUSIONS

A multidisciplinary effort to develop, disseminate, and implement an evidence-based TBI treatment protocol at a children’s hospital was associated with improved outcomes, including survival and reduced time of ICP elevation. This type of ICP-based protocol can serve as a guide for other institutions looking to reduce practice disparity in the treatment of severe TBI.

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Rohini Komarla, Bruno P. Soares, Joshua J. Chern and Sarah S. Milla

Spontaneous epidural hematoma (EDH) is a rare occurrence in patients with sickle cell disease, with a small number of cases reported. Appropriate diagnosis is critical, because rapid neurosurgical intervention may be required. This unique case illustrates clinical and MRI features of an 18-year-old woman presenting with a headache and subsequent progression to severe focal neurological symptoms. Imaging demonstrated a large EDH of mixed signal characteristics and underlying calvarial infarction, requiring emergency decompression and evacuation. A second companion case is also presented. The authors discuss proposed pathophysiology of the formation of EDHs in sickle cell anemia.

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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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Kumar Vasudevan, Ahyuda Oh, R. Shane Tubbs, David Garcia, Andrew Reisner and Joshua J. Chern

OBJECTIVE

Jackson-Pratt drains (JPDs) are commonly employed in pediatric craniofacial reconstructive surgery (CRFS) to reduce postoperative wound complications, but their risk profile remains unknown. Perioperative blood loss and volume shifts are major risks of CFRS. The goal of this study was to evaluate the risks of JPD usage in CFRS, particularly with regard to perioperative blood loss, hyponatremia, intensive care unit (ICU) length of stay, and postoperative wound complications.

METHODS

The authors performed a retrospective review of data obtained in pediatric patients who underwent CFRS at a single institution, as performed by multiple surgeons between January 2010 and December 2014. Data were gathered from patients who did and did not receive JPDs at the time of surgery. Outcome measures were compared between the JPD and no-JPD groups.

RESULTS

The overall population 179 pediatric patients: 128 who received JPDs and 51 who did not. In their analysis, the authors found no significant differences in baseline patient characteristics between the two groups. The average JPD output over the first 48 hours was 222 ± 142 ml. When examining the immediate preoperative to immediate postoperative time period, no significant differences were noted between the groups with regard to the need for blood transfusion or changes in hemoglobin, hematocrit, or serum sodium levels. These differences were also not significant when examining the 48-hour postoperative period. Finally, no significant differences in hospital length of stay, ICU length of stay, or emergency department visits at 60 days were noted between the two groups.

CONCLUSIONS

In this retrospective study, the use of JPDs in pediatric CFRS was not associated with an increased risk of serious perioperative complications, although the benefits of this practice remain unclear.