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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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Philipp Hendrix, Paul M. Foreman, Mark R. Harrigan, Winfield S. Fisher III, Nilesh A. Vyas, Robert H. Lipsky, Mingkuan Lin, Beverly C. Walters, R. Shane Tubbs, Mohammadali M. Shoja, Jean-Francois Pittet, Mali Mathru and Christoph J. Griessenauer

OBJECTIVE

Cystathionine β-synthase (CBS) is involved in homocysteine and hydrogen sulfide (H2S) metabolism. Both products have been implicated in the pathophysiology of cerebrovascular diseases. The impact of CBS polymorphisms on aneurysmal subarachnoid hemorrhage (aSAH) and its clinical sequelae is poorly understood.

METHODS

Blood samples from all patients enrolled in the CARAS (Cerebral Aneurysm Renin Angiotensin System) study were used for genetic evaluation. The CARAS study prospectively enrolled aSAH patients at 2 academic institutions in the United States from 2012 to 2015. Common CBS polymorphisms were detected using 5′exonuclease genotyping assays. Analysis of associations between CBS polymorphisms and aSAH was performed.

RESULTS

Samples from 149 aSAH patients and 50 controls were available for analysis. In multivariate logistic regression analysis, the insertion allele of the 844ins68 CBS insertion polymorphism showed a dominant effect on aSAH. The GG genotype of the CBS G/A single nucleotide polymorphism (rs234706) was independently associated with unfavorable functional outcome (modified Rankin Scale Score 3–6) at discharge and last follow-up, but not clinical vasospasm or delayed cerebral ischemia (DCI).

CONCLUSIONS

The insertion allele of the 844ins68 CBS insertion polymorphism was independently associated with aSAH while the GG genotype of rs234706 was associated with an unfavorable outcome both at discharge and last follow-up. Increased CBS activity may exert its neuroprotective effects through alteration of H2S levels, and independent of clinical vasospasm and DCI.

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Ross L. Dawkins, Joseph H. Miller, Omar I. Ramadan, Michael C. Lysek, Elizabeth N. Kuhn, Brandon G. Rocque, Michael J. Conklin, R. Shane Tubbs, Beverly C. Walters, Bonita S. Agee and Curtis J. Rozzelle

OBJECTIVE

There are many classification systems for injuries of the thoracolumbar spine. The recent Thoracolumbar Injury Classification and Severity Score (TLICS) has been shown to be a reliable tool for adult patients. The aim of this study was to assess the reliability of the TLICS system in pediatric patients. The validity of the TLICS system is assessed in a companion paper.

METHODS

The medical records of pediatric patients with acute, traumatic thoracolumbar fractures at a single Level 1 trauma center were retrospectively reviewed. A TLICS was calculated for each patient using CT and MRI, along with the neurological examination recorded in the patient’s medical record. TLICSs were compared with the type of treatment received. Five raters scored all patients separately to assess interrater reliability.

RESULTS

TLICS calculations were completed for 81 patients. The mean patient age was 10.9 years. Girls represented 51.8% of the study population, and 80% of the study patients were white. The most common mechanisms of injury were motor vehicle accidents (60.5%), falls (17.3%), and all-terrain vehicle accidents (8.6%). The mean TLICS was 3.7 ± 2.8. Surgery was the treatment of choice for 33.3% of patients. The agreement between the TLICS-suggested treatment and the actual treatment received was statistically significant (p < 0.0001). The interrater reliability of the TLICS system ranged from moderate to very good, with a Fleiss’ generalized kappa (κ) value of 0.69 for the TLICS treatment suggestion among all patients; however, interrater reliability decreased when MRI was used to contribute to the TLICS. The κ value decreased from 0.73 to 0.57 for patients with CT only vs patients with CT/MRI or MRI only, respectively (p < 0.0001). Furthermore, the agreement between suggested treatment and actual treatment was worse when MRI was used as part of injury assessment.

CONCLUSIONS

The TLICS system demonstrates good interrater reliability among physicians assessing thoracolumbar fracture treatment in pediatric patients. Physicians should be cautious when using MRI to aid in the surgical decision-making process.

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Christoph J. Griessenauer, R. Shane Tubbs, Paul M. Foreman, Michelle H. Chua, Nilesh A. Vyas, Robert H. Lipsky, Mingkuan Lin, Ramaswamy Iyer, Rishikesh Haridas, Beverly C. Walters, Salman Chaudry, Aisana Malieva, Samantha Wilkins, Mark R. Harrigan, Winfield S. Fisher III and Mohammadali M. Shoja

OBJECTIVE

Renin-angiotensin system (RAS) genetic polymorphisms are thought to play a role in cerebral aneurysm formation and rupture. The Cerebral Aneurysm Renin-Angiotensin System (CARAS) study prospectively evaluated common RAS polymorphisms and their relation to aneurysmal subarachnoid hemorrhage (aSAH).

METHODS

The CARAS study prospectively enrolled aSAH patients and controls at 2 academic centers in the United States. A blood sample was obtained from all patients for genetic evaluation and measurement of plasma angiotensin-converting enzyme (ACE) concentration. Common RAS polymorphisms were detected using 5′ exonuclease (TaqMan) genotyping assays and restriction fragment length polymorphism analysis.

RESULTS

Two hundred forty-eight patients were screened, and 149 aSAH patients and 50 controls were available for analysis. There was a recessive effect of the C allele of the angiotensinogen (AGT) C/T single-nucleotide polymorphism (SNP) (OR 1.94, 95% CI 0.912–4.12, p = 0.0853) and a dominant effect of the G allele of the angiotensin II receptor Type 2 (AT2) G/A SNP (OR 2.11, 95% CI 0.972–4.57, p = 0.0590) on aSAH that did not reach statistical significance after adjustment for potential confounders. The ACE level was significantly lower in aSAH patients with the II genotype (17.6 ± 8.0 U/L) as compared with the ID (22.5 ± 12.1 U/L) and DD genotypes (26.6 ± 14.2 U/L) (p = 0.0195).

CONCLUSIONS

The AGT C/T and AT2 G/A polymorphisms were not significantly associated with aSAH after controlling for potential confounders. However, a strong trend was identified for a dominant effect of the G allele of the AT2 G/A SNP. Downregulation of the local RAS may contribute to the formation of cerebral aneurysms and subsequent presentation with aSAH. Further studies are required to elucidate the relevant pathophysiology and its potential implication in treatment of patients with aSAH.

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R. Shane Tubbs, Andrés A. Maldonado, Yolanda Stoves, Fabian N. Fries, Rong Li, Marios Loukas, Rod J. Oskouian and Robert J. Spinner

OBJECTIVE

The accessory nerve is frequently repaired or used for nerve transfer. The length of accessory nerve available is often insufficient or marginal (under tension) for allowing direct coaptation during nerve repair or nerve transfer (neurotization), necessitating an interpositional graft. An attractive maneuver would facilitate lengthening of the accessory nerve for direct coaptation. The aim of the present study was to identify an anatomical method for such lengthening.

METHODS

In 20 adult cadavers, the C-2 or C-3 connections to the accessory nerve were identified medial to the sternocleidomastoid (SCM) muscle and the anatomy of the accessory nerve/cervical nerve fibers within the SCM was documented. The cervical nerve connections were cut. Lengths of the accessory nerve were measured. Samples of the cut C-2 and C-3 nerves were examined using immunohistochemistry.

RESULTS

The anatomy and adjacent neural connections within the SCM are complicated. However, after the accessory nerve was “detethered” from within the SCM and following transection, the additional length of the accessory nerve increased from a mean of 6 cm to a mean of 10.5 cm (increase of 4.5 cm) after cutting the C-2 connections, and from a mean of 6 cm to a mean length of 9 cm (increase of 3.5 cm) after cutting the C-3 connections. The additional length of accessory nerve even allowed direct repair of an infraclavicular target (i.e., the proximal musculocutaneous nerve). The cervical nerve connections were shown not to contain motor fibers.

CONCLUSIONS

An additional length of the accessory nerve made available in the posterior cervical triangle can facilitate direct repair or neurotization procedures, thus eliminating the need for an interpositional nerve graft, decreasing the time/distance for regeneration and potentially improving clinical outcomes.

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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.

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Ahyuda Oh, Michael Sawvel, David Heaner, Amina Bhatia, Andrew Reisner, R. Shane Tubbs and Joshua J. Chern

OBJECTIVE

Past studies have suggested correlations between abusive head trauma and concurrent cervical spine (c-spine) injury. Accordingly, c-spine MRI (cMRI) has been increasingly used in radiographic assessments. This study aimed to determine trends in cMRI use and treatment, and outcomes related to c-spine injury in children with nonaccidental trauma (NAT).

METHODS

A total of 503 patients with NAT who were treated between 2009 and 2014 at a single pediatric health care system were identified from a prospectively maintained database. Additional data on selected clinical events were retrospectively collected from electronic medical records. In 2012, a clinical pathway on cMRI usage for patients with NAT was implemented. The present study compared cMRI use and clinical outcomes between the prepathway (2009–2011) and postpathway (2012–2014) periods.

RESULTS

There were 249 patients in the prepathway and 254 in the postpathway groups. Incidences of cranial injury and Injury Severity Scores were not significantly different between the 2 groups. More patients underwent cMRI in the years after clinical pathway implementation than before (2.8% vs 33.1%, p < 0.0001). There was also a significant increase in cervical collar usage from 16.5% to 27.6% (p = 0.004), and more patients were discharged home with cervical collar immobilization. Surgical stabilization occurred in a single case in the postpathway group.

CONCLUSIONS

Heightened awareness of potential c-spine injury in this population increased the use of cMRI and cervical collar immobilization over a 6-year period. However, severe c-spine injury remains rare, and increased use of cMRI might not affect outcomes markedly.

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Przemysław A. Pękala, Brandon M. Henry, Jakub R. Pękala, Wan Chin Hsieh, Jens Vikse, Beatrice Sanna, Jerzy A. Walocha, R. Shane Tubbs and Krzysztof A. Tomaszewski

OBJECTIVE

The foramen arcuale (FA) is a bony bridge located over the vertebral artery on the posterior arch of the atlas. The presence of an FA can pose a risk during neurosurgery by providing a false impression of a broader posterior arch. The aim of this study was to provide the most comprehensive investigation on the prevalence of the FA and its clinically important anatomical features.

METHODS

Major electronic databases were searched to identify all studies that reported relevant data on the FA and the data were pooled into a meta-analysis.

RESULTS

A total of 127 studies (involving 55,985 subjects) were included. The overall pooled prevalence of a complete FA was 9.1% (95% CI 8.2%–10.1%) versus an incomplete FA, which was 13.6% (95% CI 11.2%–16.2%). The complete FA was found to be most prevalent in North Americans (11.3%) and Europeans (11.2%), and least prevalent among Asians (7.5%). In males (10.4%) the complete FA was more common than in females (7.3%) but an incomplete FA was more commonly seen in females (18.5%) than in males (16.7%). In the presence of a complete FA, a contralateral FA (complete or incomplete) was found in 53.1% of cases.

CONCLUSIONS

Surgeons should consider the risk for the presence of an FA prior to procedures on the atlas in each patient according to sex and ethnic group. We suggest preoperative screening with computerized tomography as the gold standard for detecting the presence of an FA.

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Christoph J. Griessenauer, R. Shane Tubbs, Paul M. Foreman, Michelle H. Chua, Nilesh A. Vyas, Robert H. Lipsky, Mingkuan Lin, Ramaswamy Iyer, Rishikesh Haridas, Beverly C. Walters, Salman Chaudry, Aisana Malieva, Samantha Wilkins, Mark R. Harrigan, Winfield S. Fisher III and Mohammadali M. Shoja

OBJECTIVE

Renin-angiotensin system (RAS) genetic polymorphisms are thought to play a role in cerebral aneurysm formation and rupture. The Cerebral Aneurysm Renin Angiotensin System (CARAS) study prospectively evaluated associations of common RAS polymorphisms and clinical course after aneurysmal subarachnoid hemorrhage (aSAH).

METHODS

The CARAS study prospectively enrolled aSAH patients at 2 academic centers in the United States. A blood sample was obtained from all patients for genetic evaluation and measurement of plasma angiotensin converting enzyme (ACE) concentration. Common RAS polymorphisms were detected using 5′exonuclease genotyping assays and pyrosequencing. Analysis of associations of RAS polymorphisms and clinical course after aSAH were performed.

RESULTS

A total of 166 patients were screened, and 149 aSAH patients were included for analysis. A recessive effect of allele I (insertion) of the ACE I/D (insertion/deletion) polymorphism was identified for Hunt and Hess grade in all patients (OR 2.76, 95% CI 1.17–6.50; p = 0.0206) with subsequent poor functional outcome. There was a similar effect on delayed cerebral ischemia (DCI) in patients 55 years or younger (OR 3.63, 95% CI 1.04–12.7; p = 0.0439). In patients older than 55 years, there was a recessive effect of allele A of the angiotensin II receptor Type 2 (AT2) A/C single nucleotide polymorphism (SNP) on DCI (OR 4.70, 95% CI 1.43–15.4; p = 0.0111).

CONCLUSIONS

Both the ACE I/D polymorphism and the AT2 A/C single nucleotide polymorphism were associated with an age-dependent risk of delayed cerebral ischemia, whereas only the ACE I/D polymorphism was associated with poor clinical grade at presentation. Further studies are required to elucidate the relevant pathophysiology and its potential implication in the treatment of patients with aSAH.

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Paul M. Foreman, Michelle H. Chua, Mark R. Harrigan, Winfield S. Fisher III, R. Shane Tubbs, Mohammadali M. Shoja and Christoph J. Griessenauer

OBJECTIVE

Delayed cerebral ischemia (DCI) following aneurysmal subarachnoid hemorrhage (aSAH) occurs in approximately 30% of patients. The Practical Risk Chart was developed to predict DCI based on admission characteristics; the authors seek to externally validate and critically appraise this prediction tool.

METHODS

A prospective cohort of aSAH patients was used to externally validate the previously published Practical Risk Chart. The model consists of 4 variables: clinical condition on admission, amount of cisternal and intraventricular blood on CT, and age. External validity was assessed using logistic regression. Model discrimination was evaluated using the area under the receiver operating characteristic curve (AUC).

RESULTS

In a cohort of 125 patients with aSAH, the Practical Risk Chart adequately predicted DCI, with an AUC of 0.66 (95% CI 0.55–0.77). Clinical grade on admission and amount of intracranial blood on CT were the strongest predictors of DCI and clinical vasospasm. The best-fit model used a combination of the Hunt and Hess grade and the modified Fisher scale to yield an AUC of 0.76 (95% CI 0.675–0.85) and 0.70 (95% CI 0.602–0.8) for the prediction of DCI and clinical vasospasm, respectively.

CONCLUSIONS

The Practical Risk Chart adequately predicts the risk of DCI following aSAH. However, the best-fit model represents a simpler stratification scheme, using only the Hunt and Hess grade and the modified Fisher scale, and produces a comparable AUC.