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• Journal of Neurosurgery: Pediatrics
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## Association of meningitis and clival canal defect: case illustration, management, and systematic review of the literature

### OBJECTIVE

Improper embryological development of the clivus, a bony structure that comprises part of the skull base, can lead to a clival canal defect. Previously thought to be a benign condition, clival canals have been reported to be associated with meningitis and meningoceles. In this review, the authors sought to present an unpublished case of a patient with a clival canal defect associated with meningitis and to evaluate all other reported cases.

### METHODS

In October 2020, a search of PubMed, Web of Science, and Scopus was conducted to identify all cases of clival canals reported from January 1, 1980, through October 31, 2020.

### RESULTS

Including the case presented herein, 13 cases of clival canals, 11 in children (84.6%) and 2 in adults (15.4%), have been identified. Of the pediatric patients, 5 (45.5%) had an associated meningocele, and 8 (72.7%) had meningitis. Nine of the 13 patients (69.2%) had defects that were treated surgically, 5 (38.5%) by a transnasal approach and 4 (30.8%) by a transoral approach. Two patients (15.4%) were treated with drainage and antibiotics, 1 patient (7.7%) was treated solely with antibiotics, and 1 patient (7.7%) was not treated. In the literature review, 8 reports of clival canals were found to be associated with meningitis, further contributing to the notion that the clival canal may be an overlooked source of recurrent infection. In several of these cases, surgical repair of the lesion was curative, thus preventing continued episodes of meningitis.

### CONCLUSIONS

When a patient has recurrent meningitis with no clear cause, taking a closer look at clival anatomy is recommended. In addition, if a clival canal defect has been identified, surgical repair should be considered a safe and effective primary treatment option.

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## Letter to the Editor. Entry site for ventriculoperitoneal shunt insertion and de novo post-shunt seizures

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## Surgical management of quadrigeminal cistern arachnoid cysts: case series and literature review

### OBJECTIVE

Quadrigeminal cistern arachnoid cysts (QACs) are congenital lesions that can cause pineal region compression and obstructive hydrocephalus when sufficiently large. Management of these cysts is controversial and rates of reintervention are high. Given the limited data on the management of QACs, the authors retrospectively reviewed 20 years of cases managed at their institution and performed a literature review on this topic.

### METHODS

The authors performed a retrospective analysis of patients treated for QAC at their institution between 2001 and 2021. They also performed a literature review of studies published between 1980 and 2021 that reported at least 5 patients treated for QACs. Patient characteristics, radiographic findings, management course, and postoperative follow-up data were collected and analyzed.

### RESULTS

A total of 12 patients treated for a QAC at the authors’ institution met the inclusion criteria for analysis. Median age was 9 months, mean cyst size was 5.1 cm, and 83% of patients had hydrocephalus. Initial treatment was endoscopic fenestration in 92% of these patients, 27% of whom had an endoscopic third ventriculostomy (ETV) performed concurrently. Reintervention was required in 42% of patients. Cases that required reintervention had a statistically significant lower median age at the initial intervention (5 months) than the cases that did not require reintervention (24.33 months; p = 0.018). There were no major complications. At a mean follow-up of 5.42 years, 83% of patients had improvement or resolution of their symptoms. A literature review revealed 7 studies that met the inclusion criteria, totaling 108 patients with a mean age of 8.8 years. Eighty-seven percent of patients had hydrocephalus at presentation. Ninety-two percent of patients were initially treated with endoscopic fenestration, 44% of whom underwent concurrent ETV. Complications occurred in 17.6% of cases, and reintervention was required in 30.6% of cases. The most frequent reason for reintervention was untreated or unresolved hydrocephalus after the initial procedure.

### CONCLUSIONS

Endoscopic fenestration is the most common treatment for QACs. While generally safe and effective, there is a high rate of reintervention after initial treatment of QACs, which may be associated with a younger age at the first intervention. Additionally, identifying patients who require initial treatment of hydrocephalus is critically important, as the literature suggests that untreated hydrocephalus is a common cause of reintervention.

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## Ivy sign: a diagnostic and prognostic biomarker for pediatric moyamoya

### OBJECTIVE

Ivy sign is a radiographic finding on FLAIR MRI sequences and is associated with slow cortical blood flow in moyamoya. Limited data exist on the utility of the ivy sign as a diagnostic and prognostic tool in pediatric patients, particularly outside of Asian populations. The authors aimed to investigate a modified grading scale with which to characterize the prevalence and extent of the ivy sign in children with moyamoya and evaluate its efficacy as a biomarker in predicting postoperative outcomes, including stroke risk.

### METHODS

Pre- and postoperative clinical and radiographic data of all pediatric patients (21 years of age or younger) who underwent surgery for moyamoya disease or moyamoya syndrome at two major tertiary referral centers in the US and Israel, between July 2009 and August 2019, were retrospectively reviewed. Ivy sign scores were correlated to Suzuki stage, Matsushima grade, and postoperative stroke rate to quantify the diagnostic and prognostic utility of ivy sign.

### RESULTS

A total of 171 hemispheres in 107 patients were included. The median age at the time of surgery was 9 years (range 3 months–21 years). The ivy sign was most frequently encountered in association with Suzuki stage III or IV disease in all vascular territories, including the anterior cerebral artery (53.7%), middle cerebral artery (56.3%), and posterior cerebral artery (47.5%) territories. Following surgical revascularization, 85% of hemispheres with Matsushima grade A demonstrated a concomitant, statistically significant reduction in ivy sign scores (OR 5.3, 95% CI 1.4–20.0; p = 0.013). Postoperatively, revascularized hemispheres that exhibited ivy sign score decreases had significantly lower rates of postoperative stroke (3.4%) compared with hemispheres that demonstrated no reversal of the ivy sign (16.1%) (OR 5.5, 95% CI 1.5–21.0; p = 0.008).

### CONCLUSIONS

This is the largest study to date that focuses on the role of the ivy sign in pediatric moyamoya. These data demonstrate that the ivy sign was present in approximately half the pediatric patients with moyamoya with Suzuki stage III or IV disease, when blood flow was most unstable. The authors found that reversal of the ivy sign provided both radiographic and clinical utility as a prognostic biomarker postoperatively, given the statistically significant association with both better Matsushima grades and a fivefold reduction in postoperative stroke rates. These findings can help inform clinical decision-making, and they have particular value in the pediatric population, as the ability to minimize additional radiographic evaluations and tailor radiographic surveillance is requisite.

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## Letter to the Editor. Seizures in pediatric patients with cerebral AVMs

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## Risk factors for postoperative cerebellar mutism syndrome in pediatric patients: a systematic review and meta-analysis

### OBJECTIVE

A review article assessing all the risk factors reported in the literature for postoperative cerebellar mutism syndrome (pCMS) among children remains absent. The authors sought to perform a systematic review and meta-analysis to evaluate this issue.

### METHODS

PubMed, Embase, and Web of Science were queried to systematically extract potential references. The articles relating to pCMS were required to be written in the English language, involve pediatric patients (≤ 18 years of age), and provide extractable data, which included a comparison group of patients who did not develop pCMS. The quality of the included studies was evaluated using the Newcastle-Ottawa Scale. Data were pooled using RevMan 5.4, and publication bias was assessed by visual inspection for funnel plot asymmetry. The study protocol was registered through PROSPERO (ID: CRD42021256177).

### RESULTS

Overall, 28 studies involving 2276 patients were included. Statistically significant risk factors identified from univariate analysis were brainstem invasion (OR 4.28, 95% CI 2.23–8.23; p < 0.0001), fourth ventricle invasion (OR 12.84, 95% CI 4.29–38.44; p < 0.00001), superior cerebellar peduncle invasion (OR 6.77, 95% CI 2.35–19.48; p = 0.0004), diagnosis of medulloblastoma (OR 3.26, 95% CI 1.93–5.52; p < 0.0001), medulloblastoma > 50 mm (OR 8.85, 95% CI 1.30–60.16; p = 0.03), left-handedness (OR 6.57, 95% CI 1.25–34.44; p = 0.03), and a vermis incision (OR 5.44, 95% CI 2.09–14.16; p = 0.0005). On the other hand, a tumor located in the cerebellar hemisphere (OR 0.23, 95% CI 0.06–0.92; p = 0.04), cerebellar hemisphere compression (OR 0.23, 95% CI 0.11–0.45; p < 0.0001), and intraoperative imaging (OR 0.36, 95% CI 0.18–0.72; p = 0.004) reduced the risk of pCMS.

### CONCLUSIONS

This study provides the largest and most reliable review of risk factors associated with pCMS. Although some risk factors may be dependent on one another, the data may be used by surgeons to better identify patients at risk for pCMS and for intervention planning.

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## Editorial. Social determinants in care for dysraphism

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## Estimating the prevalence of neurosurgical interventions in adults with spina bifida using the Health Facts data set: implications for transition planning and the development of adult clinics

### OBJECTIVE

As the care of patients with spina bifida continues to evolve, life expectancy is increasing, leading to a critical need for transition planning from pediatric-based to adult-based care. The burden of neurosurgical care for adults with spina bifida remains unknown. In this study, the authors sought to use a large national data set to estimate the prevalence of neurosurgical interventions in adults with spina bifida.

### METHODS

This study utilized Health Facts, which is a de-identified proprietary data set abstracted from all Cerner electronic health records. It includes 69 million unique patients with > 500 million encounters in 580 centers. Validation, technical exclusions, and data filters were applied to obtain an appropriate cohort of patients. The ICD-9 and ICD-10 codes for all types of spinal dysraphism, as well as the Current Procedural Terminology (CPT) codes for hydrocephalus procedures, spinal cord untethering, and Chiari decompression, were queried and records were retrieved. Demographic variables along with differences in age groups and temporal trends were analyzed.

### RESULTS

Overall, 24,764 unique patients with ≥ 1 encounter with a spinal dysraphism diagnosis between 2000 and 2017 were identified. The pediatric cohort included 11,123 patients with 60,027 separate encounters, and the adult cohort included 13,641 patients with 41,618 separate encounters. The proportion of females was higher in the adult (62.9%) than in the pediatric (51.4%) cohort. Annual encounters were stable from 2 to 18 years of age, but then decreased by approximately half with a precipitous drop after age 21 years. The sex distribution of adults and children who underwent procedures was similar (54.6% female adults vs 52.4% female children). Surgical interventions in adults were common. Between 2013 and 2017, there were 4913 procedures for hydrocephalus, with 2435 (49.6%) adult patients. Similarly, 273 (33.3%) of the 819 tethered cord procedures were performed in adults, as were 307 (32.9%) of 933 Chiari decompressions.

### CONCLUSIONS

The Health Facts database offered another option for studying care delivery and utilization in patients aging with spina bifida. The median age of this population has now reached early adulthood, and a significant number of neurosurgical procedures were performed in adults. An abrupt drop in the rate of encounters occurred at 21 years of age, possibly reflecting transition issues such as access-to-care problems and lack of coordinated care.

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## Importance of tranexamic acid in pediatric monosutural craniosynostosis surgery

### OBJECTIVE

Tranexamic acid (TXA) is an antifibrinolytic drug that has achieved significant reduction in perioperative blood loss and the quantity of blood transfused in many pediatric surgical procedures, without morbidity. Despite the accumulation of evidence regarding its effectiveness in craniosynostosis repair surgery, TXA is not unanimously employed by pediatric neurosurgery teams. The purpose of this study was to evaluate the impact of the routine use of TXA in a homogeneous population of children who underwent open surgery for monosutural craniosynostosis.

### METHODS

The authors retrospectively reviewed 3 cohorts of patients that were subdivided from 2 cohorts surrounding the initiation of TXA (group 1, TXA− [2008–2011] and group 2, TXA+ [2011–2013]) and a third cohort of more recent patients (group 3, TXA+ [2016–2017]). TXA was administered using the same protocol comprising a loading dose of 10 mg/kg over 15 minutes after induction of general anesthesia followed by a 10 mg/kg per hour infusion until skin closure. Patients in all 3 groups underwent similar standardized procedures for scaphocephaly, trigonocephaly, and unicoronal craniosynostosis by the same pediatric neurosurgeon.

### RESULTS

Overall, 102 infants were included in the study: 32 infants in group 1, 36 in group 2, and 34 in group 3. Significant reductions in transfusion of packed erythrocytes (PE) and fresh-frozen plasma (FFP) were observed between the TXA− and TXA+ time periods. The median volume of PE transfusion was reduced by > 50% with the use of TXA (42.8 mL/kg in the TXA− group vs 20.0 in the TXA+ groups, p < 0.0001). Reduction in PE transfusion was 100% postoperatively in the TXA+ groups (20.0 mL/kg in the TXA− group vs 0.0 in the TXA+ groups, p < 0.0001). The median volume of FFP transfusion was reduced by 100% with the use of TXA (12.8 mL/kg in the TXA− group vs 0.0 in the TXA+ groups, p < 0.0001). All children in group 1 received a transfusion, whereas 3 children (8%) and 7 children (20%) in groups 2 and 3, respectively, did not. Significant reductions in postoperative drain output were also noted between the TXA− and TXA+ time periods. The total hospital length of stay was significantly lower in the TXA+ groups (p < 0.0001).

### CONCLUSIONS

Use of TXA reduced blood loss and the need for transfusions but also decreased the hospital length of stay and, thus, minimized overall medical care costs. Intraoperative administration of TXA in craniosynostosis repair surgery should be routinely used in all centers that practice these procedures.

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## Sociodemographic disparities in fetal surgery for myelomeningocele: a single-center retrospective review

### OBJECTIVE

Fetal surgery for myelomeningocele has become an established treatment that offers less risk of requiring a ventricular shunt and improved functional outcomes for patients. An increasing body of literature has suggested that social determinants of health have a profound influence on health outcomes. The authors sought to determine the socioeconomic and racial and ethnic backgrounds of patients who were treated with fetal surgery versus those who underwent postnatal repair.

### METHODS

Demographic data, the method of myelomeningocele repair, insurance status, and zip code data for patients entered into the National Spina Bifida Patient Registry (NSBPR) from Children’s Wisconsin were collected. The zip code was used to determine the Distressed Communities Index (DCI) score, a composite socioeconomic ranking with scores ranging from 0 (no distress) to 100 (severe distress). The zip code was also used to determine the median household income for each patient based on the US Census Bureau 2013–2017 American Community Survey 5-year estimates.

### RESULTS

A total of 205 patients were identified with zip code and insurance data. There were 23 patients in the fetal surgery group and 182 patients in the postnatal surgery group. All patients were born between 2000 and 2019. Patients in the fetal surgery group were more likely to have commercial insurance (100% vs 52.2%, p < 0.001). Fetal surgery patients were also more likely to be non-Hispanic White (95.7% vs 68.7%, p = 0.058), just missing the level of statistical significance. Patients who underwent fetal surgery tended to reside in zip codes with a higher median household income (mean $66,507 vs$59,133, p = 0.122) and less-distressed communities (mean DCI score 31.3 vs 38.5, p = 0.289); however, these differences did not reach statistical significance.

### CONCLUSIONS

Patients treated with fetal surgery were more likely to have commercial insurance and have a non-Hispanic White racial and ethnic background. The preliminary data suggest that socioeconomic and racial and ethnic disparities may exist regarding access to fetal surgery, and investigation of a larger population of spina bifida patients is warranted.