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Establishing language laterality: does resting-state functional MRI help?

Sumit Singh, Mustafa S. M. Alhasan, Zhiyue Wang, Rebekah Clarke, Yin Xi, Joseph A. Maldjian, Ben Wagner, and Timothy Booth

OBJECTIVE

Task-based functional MRI (tb-fMRI) is now considered the standard, noninvasive technique in establishing language laterality in children for surgical planning. The evaluation can be limited due to several factors such as age, language barriers, and developmental and cognitive delays. Resting-state functional MRI (rs-fMRI) offers a potential path to establish language dominance without active task participation. The authors sought to compare the ability of rs-fMRI for language lateralization in the pediatric population with conventional tb-fMRI used as the gold standard.

METHODS

The authors performed a retrospective evaluation of all pediatric patients at a dedicated quaternary pediatric hospital who underwent tb-fMRI and rs-fMRI from 2019 to 2021 as part of the surgical workup for patients with seizures and brain tumors. Task-based fMRI language laterality was based on a patient’s adequate performance on one or more of the following: sentence completion, verb generation, antonym generation, or passive listening tasks. Resting-state fMRI data were postprocessed using statistical parametric mapping, FMRIB Software Library, and FreeSurfer as described in the literature. The laterality index (LI) was calculated from the independent component (IC) with the highest Jaccard Index (JI) for the language mask. Additionally, the authors visually inspected the activation maps for two ICs with the highest JIs. The rs-fMRI LI of IC1 and the authors’ image-based subjective interpretation of language lateralization were compared with tb-fMRI, which was considered the gold standard for this study.

RESULTS

A retrospective search yielded 33 patients with language fMRI data. Eight patients were excluded (5 with suboptimal tb-fMRI and 3 with suboptimal rs-fMRI data). Twenty-five patients (age range 7–19 years, male/female ratio 15:10) were included in the study. The language laterality concordance between tb-fMRI and rs-fMRI ranged from 68% to 80% for assessment based on LI of independent component analysis with highest JI and for subjective evaluation by visual inspection of activation maps, respectively.

CONCLUSIONS

The concordance rates between tb-fMRI and rs-fMRI of 68% to 80% show the limitation of rs-fMRI in determining language dominance. Resting-state fMRI should not be used as the sole method for language lateralization in clinical practice.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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Parental sensitivity and specificity to recognize shunt malfunction in their child: a single-center prospective study

Amparo Saenz, Santiago Cicutti, Romina Argañaraz, Beatriz Mantese, and Diego Giunta

OBJECTIVE

The objective of this study was to estimate the diagnostic performance (sensitivity, specificity, positive predictive value, and negative predictive value) for recognizing ventriculoperitoneal shunt (VPS) failure in the parents of patients 0–18 years of age who attended the hospital’s emergency room (ER). The second objective was to identify the factors associated with the parents’ ability to recognize the shunt blockage (true positives).

METHODS

A prospective cohort study was conducted between 2021 and 2022 including all patients 0–18 years of age who had a VPS and attended the hospital’s ER with symptoms that could correspond to VPS blockage. Parents were interviewed on admission and patients were assessed over time to discover potential VPS malfunction by surgery or follow-up. Consent was obtained from all participants.

RESULTS

Ninety-one patients were surveyed, and 59.3% showed evidence of a confirmed VPS blockage. Parental sensitivity was 66.7%, with a specificity of 21.6%. An association was found between parents who could correctly identify their child’s shunt block and the number of symptoms of shunt failure that the parent could name (OR 2.4, p < 0.05) as well as parents who reported vomiting and headache as symptoms of shunt malfunction (OR 6, p < 0.05). Parents who knew the first and last name of their primary neurosurgeon (OR 3.5, p < 0.05) also had better diagnostic sensitivity.

CONCLUSIONS

Parents who are more knowledgeable of their child’s disease, as well as parents who have good communication with their neurosurgeon, were found to have better diagnostic sensitivity.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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46th Annual Meeting The American Society of Pediatric Neurosurgeons

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 3 (Mar 2023)

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Effects of intraoperative motor evoked potential amplification following tetanic stimulation of the pudendal nerve in pediatric craniotomy

Ryota Sasaki, Kentaro Tamura, Shintaro Yamazaki, Tae Kyun Kim, Tsunenori Takatani, Hironobu Hayashi, Yasushi Motoyama, Ichiro Nakagawa, Young-Soo Park, Masahiko Kawaguchi, and Hiroyuki Nakase

OBJECTIVE

Monitoring the intraoperative motor evoked potentials (MEPs) in pediatric craniotomy is challenging because of its low detection rate, which makes it unreliable. Tetanic stimulation of the peripheral nerves of the extremities and pudendal nerves prior to transcranial electrical stimulation (TES) or direct cortical stimulation (DCS) amplifies the MEPs. The authors investigated the effects of MEP amplification following tetanic stimulation of the median and tibial nerve or the pudendal nerve in pediatric patients undergoing craniotomy.

METHODS

This prospective observational study included 15 patients ≤ 15 years of age (mean age 8.9 ± 4.9 years) undergoing craniotomy. MEPs were obtained with TES (15 cases) or DCS (8 cases)—conventional MEP without tetanic stimulation (c-MEP) and MEP following tetanic stimulation of the unilateral median and tibial nerves (mt-MEP) or following tetanic stimulation of the pudendal nerve (p-MEP) were used. Compound muscle action potentials were elicited from the abductor pollicis brevis, gastrocnemius, tibialis anterior, and abductor hallucis longus muscles. The authors compared the identification rate and the rate of amplitude increase of each MEP.

RESULTS

For both TES and DCS, the identification and amplitude increase rates were significantly higher in cases without preoperative hemiparesis for p-MEPs than in those for c-MEPs and mt-MEPs. In comparison to patients with preoperative hemiparesis, p-MEPs displayed a higher identification rate, with fewer false negatives in DCS cases.

CONCLUSIONS

In pediatric craniotomy, the authors observed the amplification effect of MEPs with pudendal nerve tetanic stimulation and the amplification effect of DCS on MEPs without increasing false negatives. These findings suggested the likelihood of more reliable intraoperative MEP monitoring in pediatric cases.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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External validation of the R2eD AVM scoring system to assess rupture risk in pediatric AVM patients

Shivani D. Rangwala, John S. Albanese, Anna L. Slingerland, Joanna E. Papadakis, Daniel S. Weber, Edward R. Smith, R. Michael Scott, and Alfred P. See

OBJECTIVE

The assessment of hemorrhagic risk is of central importance in the management of pediatric brain arteriovenous malformations (AVMs). A recently published scoring system, the R2eD AVM (race, exclusive deep location or not, AVM size < 3 or ≥ 3, venous drainage exclusively deep or not, and monoarterial feeding or >1 arterial feeders) considers patient race and AVM location, size, venous drainage, and arterial supply and was successfully externally validated for use in adults to predict the risk of AVM hemorrhagic presentation. In this study the authors sought to externally validate the R2eD AVM score for prediction of hemorrhagic risk in the pediatric AVM population.

METHODS

A pediatric database at Boston Children’s Hospital was retrospectively reviewed for all patients with a diagnosis of intracranial AVM. Exclusion criteria included age > 21 years, multiple AVMs, and incomplete clinical/radiographic data. Demographic data and R2eD AVM score calculations were collected for all patients. Univariate binary logistic regression and multivariate stepwise backward elimination models were used to determine factors associated with hemorrhagic presentation.

RESULTS

A total of 212 AVMs were identified in 212 patients with vascular anomalies from 1995 to 2021, and 144 patients met the inclusion criteria (74 [51.4%] male, 111 [77.1%] White), of whom 87 (60.4%) patients presented with rupture and 122 (84.7%) underwent resection. The mean age at surgery was 12 years (range 3 months to 20 years). The R2eD AVM scoring system was applied to each patient. The score components of size < 3 cm and exclusive deep venous drainage were significantly associated with rupture risk (p < 0.05). The complete model for the R2eD AVM score had an area under the curve of 0.671 (95% CI 0.586–0.757).

CONCLUSIONS

This study demonstrated poor external validity of the R2eD AVM score in predicting pediatric AVM rupture risk. The results suggest that future studies are warranted to determine a better scoring method to capture pediatric rupture risk, given the significant differences in clinical presentation in pediatric compared with adult AVM patients.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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Management of asymptomatic cutaneous lumbosacral findings in newborns: a neurosurgical perspective

Janelle L. Aby, Brandon G. Rocque, and Jaspreet Loyal

OBJECTIVE

The purpose of this study was to find areas of agreement among pediatric neurosurgeons with respect to the clinical management of asymptomatic newborns with a variety of lumbosacral skin findings.

METHODS

An electronic survey containing 18 clinical images and brief vignettes was sent to pediatric neurosurgeons within the American Academy of Pediatrics Section of Neurological Surgery (AAP SONS). In total, 38% (n = 21) of AAP SONS members submitted complete responses to the survey. Respondents were asked if they would advise routine care, watchful waiting, imaging, or subspecialty consultation for each clinical case. Responses were categorized into two groups: 1) watchful waiting and/or routine care, or 2) imaging and/or subspecialty consultation. Consensus was categorized as good (> 90% of responses in the same group), modest (70%–90%), and poor (< 70%). Demographic information, local factors impacting management, and experiences with local referral patterns were also collected.

RESULTS

Among the pediatric neurosurgeons within the AAP SONS network, the authors found high levels (> 90%) of agreement that subcutaneous lipomas, faun tail nevi, large skin tags, and deep/atypical lumbosacral dimples in asymptomatic newborns should prompt an imaging study. Similarly, the authors found high agreement that simple coccygeal dimples do not need imaging. The management of some types of lumbosacral vascular marks and gluteal crease deviations had poor agreement (< 70%). When imaging was recommended, there was preference for spinal MRI in most cases (67%).

CONCLUSIONS

Pediatric neurosurgeons generally agree that imaging of the spine is indicated for asymptomatic newborns with subcutaneous lipomas, faun tail nevi, large skin tags, or deep/atypical lumbar dimples (deep or atypical appearing). They also agree that imaging is unnecessary for infants with simple coccygeal dimples. There was a notable lack of consensus on the appropriate management of certain gluteal cleft deviations and cutaneous vascular marks.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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A multicenter initiative to reduce intrathecal baclofen pump surgical site infection: a Cerebral Palsy Research Network quality improvement project

Robert J. Bollo, Paul H. Gross, Brandon G. Rocque, Samuel R. Browd, Jeffrey S. Raskin, Jeffrey R. Leonard, Lama Albarqawi, Amy F. Bailes, and

OBJECTIVE

Intrathecal baclofen (ITB) therapy is an effective treatment for spasticity and dystonia in children with cerebral palsy (CP). However, ITB pump surgery is associated with one of the highest rates of surgical site infection (SSI) in medicine, leading to significant morbidity and expense. Surgical protocols have reduced the rate of SSI in children with other CNS implants, and single-center protocols have been effective in ITB surgery in pediatrics. The authors describe the first multicenter quality improvement (QI)–driven standardized protocol for ITB pump surgery in children with CP across the Cerebral Palsy Research Network (CPRN), implemented with the aim of reducing ITB-associated SSI.

METHODS

SSI was defined as a culture-positive infection, ITB pump system removal for suspected infection, or wound dehiscence with exposed hardware. Each center reported historical infection rates for at least 3 years before initiating the SSI protocol (preintervention phase). After initiation of a 13-step surgical protocol, a consecutive series of 130 patients undergoing 149 surgical procedures for ITB at four CPRN tertiary pediatric neurosurgery centers were prospectively enrolled at surgery during a 2-year study period (intervention phase). QI methodology was used, including development of a key driver diagram and tracking performance using run and control charts. The primary process measure goal was documented compliance with 80% of the protocol steps, and the primary outcome measure goal was a 20% reduction in 90-day infection rate. Patient characteristics were collected from the CPRN Research Electronic Data Capture registry, including age at surgery, BMI, Gross Motor Function Classification System level, and pattern of spasticity.

RESULTS

The aggregated preintervention 90-day ITB SSI rate was 4.9% (223 procedures) between 2014 and 2017. During the intervention phase, 136 of 149 ITB surgeries performed met inclusion criteria for analysis. The mean documented compliance rate with protocol steps was 75%, and the 90-day infection rate was 4.4%, with an average of 42 days from index surgery to infection.

CONCLUSIONS

This is the first multicenter QI initiative designed to reduce SSI in ITB surgery in children with CP. Ongoing enrollment and expansion of the protocol to other CPRN centers will facilitate identification of patient- and procedure-specific risk factors for SSI, and iterative plan-do-study-act cycles incorporating these data will further decrease the risk of SSI for ITB surgery in children.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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Cerebral pressure autoregulation and optimal cerebral perfusion pressure during neurocritical care of children with traumatic brain injury

Fartein Velle, Anders Lewén, Timothy Howells, Anders Hånell, Pelle Nilsson, and Per Enblad

OBJECTIVE

The management of cerebral perfusion pressure (CPP) is a challenge in children with traumatic brain injury (TBI) because the normal blood pressure is age dependent and the role of cerebral pressure autoregulation (CPA) is unclear. In this study, the authors aimed to examine the pressure reactivity index (PRx), CPP, optimal CPP (CPPopt), and deviations from CPPopt (ΔCPPopt) in a series of children with TBI generally and regarding age relations, temporal changes, and the influence on outcome.

METHODS

Intracranial pressure (ICP) and mean arterial pressure (MAP) monitoring data were collected during neurointensive care in 57 children who sustained a TBI and were ≤ 17 years of age. CPP, PRx, CPPopt, and ΔCPPopt (actual CPP − CPPopt) were calculated. Clinical outcomes at 6 months postinjury were dichotomized into favorable outcomes (Glasgow Outcome Scale [GOS] score 4 or 5) and unfavorable outcomes (GOS scores 1–3).

RESULTS

The median patient age was 15 (range 0.5–17) years, and the median Glasgow Coma Scale motor score at admission was 5 (range 2–5). Forty-nine (86%) of the 57 patients had favorable outcomes. For the entire group, lower PRx (better preserved CPA) was associated with a more favorable outcome (p = 0.023, ANCOVA adjusted for age). When the children were divided into age groups, this finding was statistically significant in children ≤ 15 years of age (p = 0.016), but not in children ≥ 16 years (p = 0.528). In children ≤ 15 years, a lower proportion of time with ΔCPPopt < −10% was significantly associated with a favorable outcome (p = 0.038), but not in the older age group. Temporal analysis indicated that PRx was higher (more impaired CPA) from day 4 and CPPopt was higher from day 6 in the unfavorable outcome group compared with the favorable outcome group, although those findings were not significant.

CONCLUSIONS

Impaired CPA is related to poor outcome, particularly in children ≤ 15 years of age. In that age group, actual CPP below the CPPopt level contributed significantly to unfavorable outcome, while levels close to or above the CPPopt were unrelated to outcome. CPPopt appears to be higher during the time period when CPA is most impaired.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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A comparison of incidental and symptomatic unruptured brain arteriovenous malformations in children

Alex Y. Lu, Ethan A. Winkler, Joseph H. Garcia, Kunal P. Raygor, Heather J. Fullerton, Christine K. Fox, Helen Kim, Kurtis I. Auguste, Peter P. Sun, Steven W. Hetts, Michael T. Lawton, Adib A. Abla, and Nalin Gupta

OBJECTIVE

Patients with unruptured brain arteriovenous malformations (AVMs) may present with headaches, seizures, and/or neurological deficits. A smaller number of cases may be discovered incidentally. These lesions remain incompletely understood due to their sparse reporting. Herein, the authors describe the largest series to date comparing the presentation, angioarchitecture, and management of incidental versus symptomatic unruptured AVMs in children.

METHODS

The authors performed a retrospective analysis of patients who presented with brain AVMs from 1998 to 2022 at the University of California, San Francisco. Inclusion criteria were age ≤ 18 years at the time of presentation and an angiographically proven unruptured AVM that had been diagnosed postnatally.

RESULTS

Of 76 children with unruptured AVMs, 66 (86.8%) presented with headaches, seizures, and/or neurological deficit. Ten AVMs (13.1%) were incidentally discovered through unrelated disease workup (50%), cranial trauma (40%), or research study participation (10%). Compared with patients with symptomatic unruptured AVMs, patients with incidental unruptured AVMs had a smaller mean ± SD maximum nidus diameter (2.82 ± 1.1 vs 3.98 ± 1.52 cm, p = 0.025) and fewer had deep venous drainage (20% of patients vs 61%, p = 0.036). They also presented at an earlier age (10 ± 5.2 vs 13.5 ± 4 years, p = 0.043) and with longer duration to first treatment (541 ± 922 vs 196 ± 448 days, p = 0.005). During the observation period, 1 patient developed recurring headaches and demonstrated AVM nidus growth. Four AVMs greater than 3 cm in size or in a deep location were treated with radiosurgery. Six other AVMs were treated with resection, with 2 receiving preoperative embolization. Eight AVMs (80%) were obliterated on last follow-up. Postprocedural complications included 2 transient neurological deficits after resection and 1 case of delayed seizure development after radiosurgery. The mean follow-up period was 5.7 ± 5.7 years without any hemorrhage episodes.

CONCLUSIONS

A substantial proportion of pediatric patients with unruptured AVMs are discovered incidentally. With earlier presentation and more elementary angioarchitecture than symptomatic unruptured AVMs, these incidental lesions provide a snapshot into the natural history of AVM before symptom development or rupture.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)

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Comparison of neurocognitive and quality-of-life outcomes in pediatric craniopharyngioma patients treated with partial resection and radiotherapy versus gross-total resection only

Guillermo Aldave, M. Fatih Okcu, Murali Chintagumpala, Lucia Ruggieri, Charles G. Minard, Fatema Malbari, Lisa E. Mash, Arnold C. Paulino, Susan McGovern, Uma Ramaswamy, William Whitehead, and Lisa S. Kahalley

OBJECTIVE

The optimal management of pediatric craniopharyngioma patients remains controversial, shifting from radical resection (gross-total resection [GTR]) to a more conservative approach with partial resection/biopsy followed by radiotherapy (PR+RT). To the authors’ knowledge, no previous studies have compared neurocognitive and quality-of-life (QOL) outcomes between the two main treatments. In this study, the authors compared changes in intellectual, adaptive, and QOL scores in children treated for craniopharyngioma with GTR and those treated with PR+RT.

METHODS

Patients underwent annual neurocognitive and QOL evaluations for up to 10 years posttreatment, including the Full-Scale IQ, Verbal Comprehension Index (VCI), Perceptual Reasoning Index (PRI), Working Memory Index (WMI), and Processing Speed Index (PSI). Child- and parent-reported QOL scores and adaptive behavior in different domains were assessed. General linear mixed models were used to examine change in scores over time by treatment group with adjustment for significant covariates.

RESULTS

Scores from 43 patients treated between 2009 and 2019 (21 GTR, 22 PR+RT) were examined. Within the PR+RT group, 9 patients had intensity-modulated RT and 13 had proton beam therapy. The treatment groups were similar in sex (44% male) and age (median 7.3 years). There were no significant differences in the trajectory of intellectual functioning or QOL scale scores between the two groups. However, patients who underwent GTR exhibited significant improvement over time in overall adaptive behavior (p = 0.04) and conceptual skills (p = 0.01), which was not observed in patients treated with PR+RT.

CONCLUSIONS

Long-term pediatric craniopharyngioma survivors treated with GTR and PR+RT have similar intellectual function and QOL. Larger studies are needed to explore small but clinically significant differences between the two groups.

In Journal of Neurosurgery: Pediatrics Volume 31: Issue 5 (May 2023)