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Patrick W. Hullett, Nazineen Kandahari, Tina T. Shih, Jonathan K. Kleen, Robert C. Knowlton, Vikram R. Rao, and Edward F. Chang

BACKGROUND

In classic speech network models, the primary auditory cortex is the source of auditory input to Wernicke’s area in the posterior superior temporal gyrus (pSTG). Because resection of the primary auditory cortex in the dominant hemisphere removes inputs to the pSTG, there is a risk of speech impairment. However, recent research has shown the existence of other, nonprimary auditory cortex inputs to the pSTG, potentially reducing the risk of primary auditory cortex resection in the dominant hemisphere.

OBSERVATIONS

Here, the authors present a clinical case of a woman with severe medically refractory epilepsy with a lesional epileptic focus in the left (dominant) Heschl’s gyrus. Analysis of neural responses to speech stimuli was consistent with primary auditory cortex localization to Heschl’s gyrus. Although the primary auditory cortex was within the proposed resection margins, she underwent lesionectomy with total resection of Heschl’s gyrus. Postoperatively, she had no speech deficits and her seizures were fully controlled.

LESSONS

While resection of the dominant hemisphere Heschl’s gyrus/primary auditory cortex warrants caution, this case illustrates the ability to resect the primary auditory cortex without speech impairment and supports recent models of multiple parallel inputs to the pSTG.

Open access

Jaime R. Guerrero, Khaled M. Taghlabi, Sara A. Meyer, Lokeshwar S. Bhenderu, Saeed S. Sadrameli, Clive I. Shkedy, Amir H. Faraji, and Robert C. Rostomily

BACKGROUND

Metastatic cancer may involve the central and peripheral nervous system, usually in the late stages of disease. At this point, most patients have been diagnosed and treated for widespread systemic disease. Rarely is the involvement of the peripheral nervous system the presenting manifestation of malignancy. One reason for this is a proposed “blood-nerve barrier” that renders the nerve sheath a relatively privileged site for metastases.

OBSERVATIONS

The authors presented a novel case of metastatic melanoma presenting as intractable leg pain and numbness. Further workup revealed concurrent disease in the brain and breast, prompting urgent treatment with radiation and targeted immunotherapy.

LESSONS

This case highlights the rare presentation of metastatic melanoma as a mononeuropathy. Although neurological complications of metastases tend to occur in later stages of disease after initial diagnosis and treatment, one must remember to consider malignancy in the initial differential diagnosis of mononeuropathy.

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Matthew Muir, Ron Gadot, Sarah Prinsloo, Hayley Michener, Jeffrey Traylor, Prazwal Athukuri, Sudhakar Tummala, Vinodh A. Kumar, and Sujit S. Prabhu

OBJECTIVE

Robust preoperative imaging can improve the extent of resection in patients with brain tumors while minimizing postoperative neurological morbidity. Both structural and functional imaging techniques can provide helpful preoperative information. A recent study found that transcranial magnetic stimulation (TMS) tractography has significant predictive value for permanent deficits. The present study directly compares the predictive value of TMS tractography and task-based functional MRI (fMRI) tractography in the same cohort of glioma patients.

METHODS

Clinical outcome data were collected from charts of patients with motor eloquent glioma and preoperative fMRI and TMS studies. The primary outcome was a new or worsened motor deficit present at the 3-month postoperative follow-up, which was termed a "permanent deficit." Postoperative MR images were overlaid onto preoperative plans to determine which imaging features were resected. Multiple fractional anisotropic thresholds (FATs) were screened for both TMS and fMRI tractography. The predictive value of the various thresholds was modeled using receiver operating characteristic curve analysis.

RESULTS

Forty patients were included in this study. Six patients (15%) sustained permanent postoperative motor deficits. A significantly greater predictive value was found for TMS tractography than for fMRI tractography regardless of the FAT. Despite 35% of patients showing clinically relevant neuroplasticity captured by TMS, only 2.5% of patients showed a blood oxygen level–dependent signal displaced from the precentral gyrus. Comparing the best-performing FAT for both modalities, TMS seeded tractography showed superior predictive value across all metrics: sensitivity, specificity, positive predictive value, and negative predictive value.

CONCLUSIONS

The results from this study indicate that the prediction of permanent deficits with TMS tractography is superior to that with fMRI tractography, possibly because TMS tractography captures clinically relevant neuroplasticity. However, future large-scale prospective studies are needed to fully illuminate the proper role of each modality in comprehensive presurgical workups for patients with motor-eloquent tumors.

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Gabriella Pendola, George W. Koutsouras, Joseph Piatt, Bruce A. Kaufman, Carolina Sandoval-Garcia, and Annie I. Drapeau

OBJECTIVE

Quality improvement (QI) is a methodology used to implement sustainable, meaningful change to improve patient outcomes. Given the complex pathologies observed in pediatric neurosurgery, QI projects could potentially improve patient care. Overall, there is a need to characterize the degree of QI opportunities, training, and initiatives within the field of pediatric neurosurgery. Herein the authors aimed to define the current QI landscape in pediatric neurosurgery.

METHODS

A cross-sectional survey was sent to all members of the American Association of Neurological Surgeons/Congress of Neurological Surgeons Joint Section on Pediatric Neurological Surgery via email. The responses were anonymized. Questions addressed several relatable QI topics including 1) training and participation in QI; 2) QI infrastructure; 3) QI program incentives; and 4) general opinions on the National Surgical Quality Improvement Program (NSQIP) database, various QI topics, and QI productivity.

RESULTS

Responses were received from 129 participants (20% response rate). Most respondents practiced in an academic setting (59.8%) and at a free-standing pediatric hospital (65.4%). Participation in QI projects was high (81.7%), but only 23.8% of respondents had formal QI training. Only 36.5% of respondents had institutional requirements for QI work; the majority of those were only required to participate as a project team member. Nearly half of the respondents did not receive incentives or institutional support for QI. The majority agreed ("strongly" and "somewhat") that a QI course would be beneficial (75.5%), that QI projects should be considered for publication in neurosurgery journals (88.1%), and that there is a need for national quality metrics (81.4%). Over 88% have an interest in seeing QI project presentations at the annual Pediatric Joint Section meeting. Only 26.3% believed that the NSQIP was a useful QI guide. Respondents suggested further study of the following QI topics: overall rates of infection and their prevention, hydrocephalus, standardized treatment algorithms for common disorders, team communication, pediatric neurosurgery–specific database, access to care, and interprofessional education.

CONCLUSIONS

Areas of opportunity include specialty-specific QI education, tactics for obtaining support to build the QI infrastructure, increased visibility of QI work within pediatric neurosurgery, and a review of available registries to provide readily available data relevant to this specialty.

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Matthew E. Pontell, Aaron M. Yengo-Kahn, Emily Taylor, Morgan Kane, J Michael Newton, Kelly A. Bennett, John C. Wellons III, and Stephane A. Braun

OBJECTIVE

The objective of this study was to determine the effects of in utero bipedicle flaps on maternal-fetal morbidity/mortality, the need for CSF diversion, and long-term functional outcomes.

METHODS

Eighty-six patients who underwent fetal myelomeningocele repair from 2011 to 2021 at a single institution were reviewed. Primary outcomes included intrauterine fetal demise, postnatal death, postnatal myelomeningocele repair dehiscence, and CSF diversion by final follow-up.

RESULTS

The cohorts were no different with regard to race, ethnicity, maternal age at fetal surgery, body mass index, gravidity, parity, gestational age at fetal surgery, estimated fetal weight at fetal surgery, or fetal lesion level. Of the 86 patients, 64 underwent primary linear repair and 22 underwent bipedicle flap repair. There were no significant differences in rates of intrauterine fetal demise, postnatal mortality, midline repair site dehiscence, or the need for CSF diversion by final follow-up. Operative times were longer (32.5 vs 18.7 minutes, p < 0.001) and gestational age at delivery was lower (232 vs 241 days, p = 0.01) in the bipedicle flap cohort, but long-term functional outcomes were not different.

CONCLUSIONS

Analysis of the total cohort affirms the long-term benefits of fetal myelomeningocele repair. In utero bipedicle flaps are safe and can be used for high-tension lesions without increasing perioperative risks to the mother or fetus. In utero flaps preserve the long-term benefits seen with primary linear repair and may expand inclusion criteria for fetal repair, providing life-changing care for more patients.

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John W. Gilbert, Brydon Christensen, and Sherri Matheny

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Douglas L. Brockmeyer, Samuel H. Cheshier, Jeff Stevens, Julio C. Facelli, Kerry Rowe, John D. Heiss, Anthony Musolf, David H. Viskochil, Kristina L. Allen-Brady, and Lisa A. Cannon-Albright

OBJECTIVE

Inherited variants predisposing patients to type 1 or 1.5 Chiari malformation (CM) have been hypothesized but have proven difficult to confirm. The authors used a unique high-risk pedigree population resource and approach to identify rare candidate variants that likely predispose individuals to CM and protein structure prediction tools to identify pathogenicity mechanisms.

METHODS

By using the Utah Population Database, the authors identified pedigrees with significantly increased numbers of members with CM diagnosis. From a separate DNA biorepository of 451 samples from CM patients and families, 32 CM patients belonging to 1 or more of 24 high-risk Chiari pedigrees were identified. Two high-risk pedigrees had 3 CM-affected relatives, and 22 pedigrees had 2 CM-affected relatives. To identify rare candidate predisposition gene variants, whole-exome sequence data from these 32 CM patients belonging to 24 CM-affected related pairs from high-risk pedigrees were analyzed. The I-TASSER package for protein structure prediction was used to predict the structures of both the wild-type and mutant proteins found here.

RESULTS

Sequence analysis of the 24 affected relative pairs identified 38 rare candidate Chiari predisposition gene variants that were shared by at least 1 CM-affected pair from a high-risk pedigree. The authors found a candidate variant in HOXC4 that was shared by 2 CM-affected patients in 2 independent pedigrees. All 4 of these CM cases, 2 in each pedigree, exhibited a specific craniocervical bony phenotype defined by a clivoaxial angle less than 125°. The protein structure prediction results suggested that the mutation considered here may reduce the binding affinity of HOXC4 to DNA.

CONCLUSIONS

Analysis of unique and powerful Utah genetic resources allowed identification of 38 strong candidate CM predisposition gene variants. These variants should be pursued in independent populations. One of the candidates, a rare HOXC4 variant, was identified in 2 high-risk CM pedigrees, with this variant possibly predisposing patients to a Chiari phenotype with craniocervical kyphosis.

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Pierre-Yves Borius, Anne Christine Januel, Jean Yves Plas, Pierre Duthil, Jean Albert Lotterie, Igor Latorzeff, and Jean Sabatier

OBJECTIVE

Dosimetric radiosurgery incidents are rare and probably insufficiently reported in scientific publications. After a long follow-up (FU), the authors studied the outcomes of patients treated with overexposure radiation for arteriovenous malformation (AVM) administered via stereotactic radiosurgery (SRS) at their department.

METHODS

Between May 2006 and June 2007, 22 patients were treated for AVM with SRS. The mean (range) patient age was 43.5 (11.8–78) years. Previous treatments were embolization (n = 10), SRS (1), and surgery (1). The average (range) volume was 2.1 (0.2–6.4) cm3. The median prescribed minimal dose was 18.0 Gy. An initial error in the estimation of scatter factors led to overexposure to radiation. Due to this incident, the median delivered minimum dose was 25.0 Gy. All patients were prospectively followed with clinical examination and imaging.

RESULTS

The mean (range) clinical FU was 14.5 (12.0–15.2) years. AVM obliteration after SRS was completed in 90.9% of patients at a mean (range) of 39.4 (24.4–70.4) months. No patient had post-SRS AVM bleeding. Three patients (13.6%) had new permanent deficits due to radiation-induced changes (RICs). Obliteration without new deficits was achieved in 18 patients (81.8%). Two patients had new epilepsy that was probably due to RIC but well controlled. The median (range) MRI FU was 13.8 (2.5–14.9) years. During MRI FU, two RIC periods were observed: one classic period during the first 3 years showed T1-weighted annular irregular enhancement (13%), and the other period between 5 and 15 years after SRS showed the occurrence of cystic and hemorrhagic lesions (22.7%). There were no cases of radiation-induced tumor.

CONCLUSIONS

The present long-term report showed that this overexposure incident probably increased the AVM obliteration rate. This overexposure seems to have induced RIC and in particular a higher rate of cystic and hemorrhagic late lesions with nevertheless moderate clinical consequences. Long-term FU for AVM is mandatory due to the risk of late RIC.

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Rosemary T. Behmer Hansen, Ryan A. Behmer Hansen, Justin L. Gold, Sai Batchu, Rebecca D. Lozada, Samantha D. Palma, Stephen J. Susman, William A. Blocher III, and Angela M. Richardson

OBJECTIVE

This study was performed to compare authorship trends, by gender, in the neurosurgical oncology literature.

METHODS

Complete author listings for neurosurgical oncology articles published between 1944 and 2021 in five top neuro-oncology journals were extracted from the PubMed database and journal websites on December 2, 2021. Author gender was characterized with the web application programming interface (API) genderize.io. The statistical significance (p < 0.05) of time-, journal-, and gender-based differences was determined by independent-samples t-test, chi-square test, and/or Fisher’s exact test.

RESULTS

A total of 14,020 articles were written by 67,115 unique authors occupying 97,418 authorship spots. The gender for 80,030 authorship positions (82.2%) was successfully characterized. Male authors were significantly more likely than the female authors to have a first-author publication, have a last-author publication, and have authored multiple articles within the data set. Among authors who published in multiple different years (n = 11,532), women had a shorter time window of publishing (5.46 vs 6.75 years between first and last publication: mean difference [MD] 1.28 [95% CI 1.06–1.50] years, p < 0.001). During this window, however, they were slightly more productive than the men, based on the mean number of publications per year (1.06 vs 1.01 articles: MD 0.05 [95% CI 0.02–0.09] articles, p = 0.002). The percentage of female authors on each neuro-oncology research team has increased by 3.3% (95% CI 2.6%–3.9%) per decade to a mean of 26.5% in the 2020s. Having a female last author was positively associated with having a female first author (OR 2.57 [95% CI 2.29–2.89]) and a higher proportion of women on the research team overall. The percentages of female first and last authors increased at significantly higher rates in medically oriented journals than in surgically oriented journals (first authors: 0.72% [95% CI 0.58%–0.87%] vs 0.36% [95% CI 0.30%–0.42%] per year, p < 0.001; and last authors: 0.50% [95% CI 0.38%–0.62%] vs −0.03% [95% CI −0.10% to 0.05%] per year, p < 0.001).

CONCLUSIONS

Female authorship in top neuro-oncology journals has increased since the 1940s, with female-led teams showing greater gender diversity. However, female researchers lag behind their male counterparts in quantity of published research and are less likely to hold first or last authorship positions. This difference is more pronounced in the three neurosurgical oncology journals than in the two medical neuro-oncology journals, which may reflect the relatively low female representation in neurosurgery relative to medical oncology. Collectively, these trends have meaningful implications for career advancement, which is often dependent on academic productivity.

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Samuel Berchi Kankam, Amin Tavallaii, Esmaeil Mohammadi, Amirhosein Nejat, Zohreh Habibi, and Farideh Nejat

OBJECTIVE

The overall prognosis of encephalocele (EC) is not well described. However, the presence of some risk factors may result in neurodevelopmental delay (NDD) and negatively affect the prognosis of affected patients. The goal of this study was to evaluate neurodevelopmental outcome, as well as the impact of a number of factors on the outcome in patients with ECs.

METHODS

This was an observational, retrospective study including 102 children with EC who were followed at the pediatric neurosurgery department of a tertiary medical center between the years 2010 and 2021. The authors evaluated NDD status according to the Centers for Disease Control and Prevention classification via clinical evaluation and parent interviews in the outpatient setting.

RESULTS

There were 52 boys and 50 girls. The median age at the time of surgery was 4 months (range 1 day–7.5 years). Seventy-one patients (69.6%) had posterior ECs, whereas 31 (30.4%) had anterior ECs. Forty-three (42.2%) of the ECs contained neural tissue. Of the 102 patients, 33 (32.4%) had ventriculomegaly. In terms of NDD, 14 (14.9%) had mild/moderate delay, whereas 17 patients (18.1%) had severe NDD. On univariate analysis, posterior location, size of sac, presence of neural tissue, ventriculomegaly, symptomatic hydrocephalus, and postoperative infection were correlated with NDD. On a multivariate logistic regression model, only neural tissue presence had a statistically significant association with NDD (OR 7.04, 95% CI 1.33–37.2, p = 0.022). Although not statistically significant, children with ventriculomegaly were 2.6 times as likely to have NDD (95% CI 0.59–11.19, p = 0.362).

CONCLUSIONS

This is a single-center study with a large sample size in which the neurodevelopmental status of patients with EC was assessed, and the authors tried to find the risk factors of NDD in these patients. The results showed that the presence of neural tissue within the EC sac was the only risk factor that had independent statistically significant association with NDD.