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Ricardo A. Domingo, Gaetano De Biase, Ramon Navarro, Jaime L. Martinez Santos, Gabriella A. Rivas, Vivek Gupta, David Miller, Bernard R. Bendok, Waleed Brinjikji, W. Christopher Fox, Thien J. Huynh, and Rabih G. Tawk

OBJECTIVE

Available data on management of sacral arteriovenous fistulas (sAVFs) are limited to individual case reports and small series. Management includes observation, endovascular embolization, or surgical ligation, with no clear guidelines on the optimal treatment modality. The authors’ objective was to report their multiinstitutional experience with management of sAVF patients, including clinical and radiographic characteristics and postprocedural outcomes.

METHODS

The electronic medical records of patients with a diagnosis of spinal arteriovenous fistula treated from January 2004 to December 2019 at the authors’ institutions were reviewed, and data were summarized using descriptive statistics, including percentage and count for categorical data, median as a measure of central tendency for continuous variables, and interquartile range (IQR) as a measure of dispersion.

RESULTS

A total of 26 patients with sAVFs were included. The median (IQR) age was 65 (57–73) years, and 73% (n = 19) of patients were male. Lower-extremity weakness was the most common presenting symptom (n = 24 [92%]), and half the patients (n = 13 [50%]) reported bowel and bladder sphincter dysfunction. The median (IQR) time from symptom onset to treatment was 12 (5.25–26.25) months. Radiographically, all patients had T2 hyperintensity at the level of the conus medullaris (CM) (n = 26 [100%]). Intradural flow voids were identified in 85% (n = 22) of patients. The majority of the lesions had a single identifiable arterial feeder (n = 19 [73%]). The fistula was located most commonly at the S1 level (n = 13 [50%]). The site where the draining vein connects to the pial venous plexus was seen predominantly at the lumbar level (n = 16 [62%]). In total, 29 procedures were performed: 10 open surgeries and 19 endovascular embolization procedures. Complete occlusion was achieved in 90% (n = 9) of patients after open surgery and 79% (n = 15) after endovascular embolization. Motor improvement was seen in 68% of patients (n = 15), and bladder and bowel function improved in 9 patients (41%). At last follow-up, 73% (n = 16) of patients had either resolution or improvement of the pretreatment intramedullary T2 signal hyperintensity.

CONCLUSIONS

T2 hyperintensity of the CM and a dilated filum terminale vein are consistent radiographic signs of sAVF, and delayed presentation is common. Complete occlusion was achieved in almost all patients after surgery, and endovascular embolization was effective in 70% of the patients. Further studies are needed to determine the best treatment modality based on case-specific characteristics.

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Victor M. Lu, Shelly Wang, David J. Daniels, Robert J. Spinner, Allan D. Levi, and Toba N. Niazi

OBJECTIVE

Malignant peripheral nerve sheath tumors (MPNSTs) are rare tumors found throughout the body, with their clinical course in children still not completely understood. Correspondingly, this study aimed to determine survival outcomes and specific clinical predictors of survival in this population from a large national database.

METHODS

All patients with MPNSTs aged ≤ 18 years in the US National Cancer Database (NCDB) between 2005 and 2016 were retrospectively reviewed. Data were summarized, and overall survival was modeled using Kaplan-Meier and Cox regression analyses.

RESULTS

A total of 251 pediatric patients with MPNSTs (132 [53%] females and 119 [47%] males) were identified; the mean age at diagnosis was 13.1 years (range 1–18 years). There were 84 (33%) MPNSTs located in the extremities, 127 (51%) were smaller than 1 cm, and 22 (9%) had metastasis at the time of diagnosis. In terms of treatment, surgery was pursued in 187 patients (74%), chemotherapy in 116 patients (46%), and radiation therapy in 129 patients (61%). The 5-year overall survival rate was estimated at 52% (95% CI 45%–59%), with a median survival of 64 months (range 36–136 months). Multivariate regression revealed that older age (HR 1.10, p < 0.01), metastases at the time of diagnosis (HR 2.14, p = 0.01), and undergoing biopsy only (HR 2.98, p < 0.01) significantly and independently predicted a shorter overall survival. Chemotherapy and radiation therapy were not statistically significant.

CONCLUSIONS

In this study, the authors found that older patient age, tumor metastases at the time of diagnosis, and undergoing only biopsy significantly and independently predicted poorer outcomes. Only approximately half of patients survived to 5 years. These results have shown a clear survival benefit in pursuing maximal safe resection in pediatric patients with MPNSTs. As such, judicious workup with meticulous resection by an expert team should be considered the standard of care for these tumors in children.

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Joseph Driver and Michael W. Groff

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Maria R. H. Castro, Stephen T. Magill, Ramin A. Morshed, Jacob S. Young, Steve E. Braunstein, Michael W. McDermott, and Edward F. Chang

OBJECTIVE

Tumors compressing the trigeminal nerve can cause facial pain, numbness, or paresthesias. Limited data exist describing how these symptoms change after resection and what factors predict symptom improvement. The objective of this study was to report trigeminal pain and sensory outcomes after tumor resection and identify factors predicting postoperative symptom improvement.

METHODS

This retrospective study included patients with tumors causing facial pain, numbness, or paresthesias who underwent resection. Trigeminal schwannomas were excluded. Logistic regression, recursive partitioning, and time-to-event analyses were used to report outcomes and identify variables associated with facial sensory outcomes.

RESULTS

Eighty-six patients met inclusion criteria, and the median follow-up was 3.1 years; 63 patients (73%) had meningiomas and 23 (27%) had vestibular schwannomas (VSs). Meningioma patients presented with pain, numbness, and paresthesias in 56%, 76%, and 25% of cases, respectively, compared with 9%, 91%, and 39%, respectively, for patients with VS. Most meningioma patients had symptoms for less than 1 year (60%), whereas the majority of VS patients had symptoms for 1–5 years (59%). The median meningioma and VS diameters were 3.0 and 3.4 cm, respectively. For patients with meningiomas, gross-total resection (GTR) was achieved in 27% of patients, near-total resection (NTR) in 29%, and subtotal resection (STR) in 44%. For patients with VS, GTR was achieved in 9%, NTR in 30%, and STR in 61%. Pain improved immediately after tumor resection in 81% of patients and in 92% of patients by 6 weeks. Paresthesias improved immediately in 80% of patients, increasing to 84% by 6 weeks. Numbness improved more slowly, with 52% of patients improving immediately, increasing to 79% by 2 years. Pain recurred in 22% of patients with meningiomas and 0% of patients with VSs. After resection, the Barrow Neurological Institute (BNI) facial pain intensity score improved in 73% of patients. The tumor diameter significantly predicted improvement in BNI score (OR 0.47/cm larger, 95% CI 0.22–0.99; p = 0.047). Complete decompression of the trigeminal nerve was associated with qualitative improvement in pain (p = 0.037) and decreased pain recurrence (OR 0.08, 95% CI 0.01–0.67; p = 0.024).

CONCLUSIONS

Most patients with facial sensory symptoms caused by meningiomas or VSs experienced improvement after resection. Surgery led to immediate and sustained improvement in pain and paresthesias, whereas numbness was slower to improve. Patients with smaller tumors and complete decompression of the trigeminal nerve were more likely to experience improvement in facial pain.

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Mohammed Ali Alvi, Archis R. Bhandarkar, David J. Daniels, Kai J. Miller, and Edward S. Ahn

OBJECTIVE

CSF shunt insertion is the most commonly performed neurosurgical procedure for pediatric patients with hydrocephalus, and complications including infections and catheter obstruction are common. The rate of readmission in the first 30 days after surgery has been used across surgical disciplines to determine healthcare quality. In the current study, the authors sought to assess factors associated with early shunt revision within 30 days using real-world data.

METHODS

Targeted shunt data set participant user files of the National Surgical Quality Improvement Program (NSQIP) from 2016 to 2019 were queried for patients undergoing a shunt procedure. A multivariable logistic regression model was performed to assess the impact of demographics, etiologies, comorbidities, congenital malformations, and shunt adjuncts on shunt revision within 30 days, as well as shunt revision due to infection within 30 days.

RESULTS

A total of 3919 primary pediatric shunt insertions were identified in the NSQIP database, with a mean (± SD) patient age of 26.3 ± 51.6 months. There were a total of 285 (7.3%) unplanned shunt revisions within 30 days, with a mean duration of 14.9 ± 8.5 days to first intervention. The most common reason for intervention was mechanical shunt failure (32.6% of revision, 2.4% overall, n = 93), followed by infection (31.2% of all interventions, 2.3% overall, n = 89) and wound disruption or CSF leak (22.1% of all interventions, 1.6% overall, n = 63). Patients younger than 6 months of age had the highest overall unplanned 30-day revision rate (8.5%, 203/2402) as well as the highest 30-day shunt infection rate (3%, 72/2402). Patients who required a revision were also more likely to have a cardiac risk factor (34.7%, n = 99, vs 29.2%, n = 1061; p = 0.048). Multivariable logistic regression revealed that compared to patients 9–18 years old, those aged 2–9 years had significantly lower odds of repeat shunt intervention (p = 0.047), while certain etiologies including congenital hydrocephalus (p = 0.0127), intraventricular hemorrhage (IVH) of prematurity (p = 0.0173), neoplasm (p = 0.0005), infection (p = 0.0004), and syndromic etiology (p = 0.0136), as well as presence of ostomy (p = 0.0095), were associated with higher odds of repeat intervention. For shunt infection, IVH of prematurity was found to be associated with significantly higher odds (p = 0.0427) of shunt infection within 30 days, while use of intraventricular antibiotics was associated with significantly lower odds (p = 0.0085).

CONCLUSIONS

In this study of outcomes after pediatric shunt placement using a nationally derived cohort, early shunt failure and infection within 30 days were found to remain as considerable risks. The analysis of this national surgical quality registry confirms that, in accordance with other multicenter studies, hydrocephalus etiology, age, and presence of ostomy are important predictors of the need for early shunt revision. IVH of prematurity is associated with early infections while intraventricular antibiotics may be protective. These findings could be used for benchmarking in hospital efforts to improve quality of care for pediatric patients with hydrocephalus.

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Robert J. Morecraft, Kimberly S. Stilwell-Morecraft, Jizhi Ge, Alexander Kraskov, and Roger N. Lemon

OBJECTIVE

In some cases of incomplete cervical spinal cord injury (iSCI) there is marked paresis and dysfunction of upper-extremity movement but not lower-extremity movement. A continued explanation of such symptoms is a somatotopic organization of corticospinal tract (CST) fibers passing through the decussation at the craniovertebral junction (CVJ) and lateral CST (LCST). In central cord syndrome, it has been suggested that injury to the core of the cervical cord may include selective damage to medially located arm/hand LCST fibers, without compromising laterally located leg fibers. Because such somatotopic organization in the primate CST might contribute to the disproportionate motor deficits after some forms of iSCI, the authors made a systematic investigation of CST organization in the CVJ and LCST using modern neuroanatomical techniques.

METHODS

High-resolution anterograde tracers were used in 11 rhesus macaque monkeys to define the course of the corticospinal projection (CSP) through the CVJ and LCST from the arm/hand, shoulder, and leg areas of the primary motor cortex (M1). This approach labels CST fibers of all sizes, large and small, arising in these areas. The CSP from the dorsolateral and ventrolateral premotor cortex and supplementary motor area were also studied. A stereological approach was adapted to quantify labeled fiber distribution in 8 cases.

RESULTS

There was no evidence for somatotopic organization of CST fibers passing through the CVJ or contralateral LCST. Fiber labeling from each cortical representation was widespread throughout the CST at the CVJ and LCST and overlapped extensively with fibers from other representations. This study demonstrated no significant difference between medial versus lateral subsectors of the LCST in terms of number of fibers labeled from the M1 arm/hand area.

CONCLUSIONS

This investigation firmly rejects the concept of somatotopy among CST fibers passing through the CVJ and LCST, in contrast with the somatotopy in the cortex, corona radiata, and internal capsule. All CST fibers in the CVJ and LCST would thus appear to be equally susceptible to focal or diffuse injury, regardless of their cortical origin. The disproportionate impairment of arm/hand movement after iSCI must therefore be due to other factors, including greater dependence of hand/arm movements on the CST compared with the lower limb. The dispersed and intermingled nature of frontomotor fibers may be important in motor recovery after cervical iSCI.

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Mohamed A. R. Soliman, Asham Khan, John Pollina, and Jeffrey P. Mullin

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Noor H. Maria and Qurrat A. Siddiq

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Paula Alcazar and Juan Casado Pellejero