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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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Nikhil K. Murthy, Kimberly K. Amrami, Stephen M. Broski, Patrick B. Johnston, and Robert J. Spinner

OBJECTIVE

Neurolymphomatosis (NL) is a rare manifestation of lymphoma confined to the peripheral nervous system that is poorly understood. It can be found in the cauda equina, but extraspinal disease can be underappreciated. The authors describe how extraspinal NL progresses to the cauda equina by perineural spread and the implications of this on timely and safe diagnostic options.

METHODS

The authors used the Mayo Clinic medical records database to find cases of cauda equina NL with sufficient imaging to characterize the lumbosacral plexus diagnosed from tissue biopsy. Demographics (sex, age), clinical data (initial symptoms, cerebrospinal fluid, evidence of CNS involvement, biopsy location, primary or secondary disease), and imaging findings were reviewed.

RESULTS

Ten patients met inclusion and exclusion criteria, and only 2 of 10 patients presented with cauda equina symptoms at the time of biopsy, with 1 patient undergoing a cauda equina biopsy. Eight patients were diagnosed with diffuse large B-cell lymphoma, 1 with low-grade B-cell lymphoma, and 1 with mantle cell lymphoma. Isolated spinal nerve involvement was identified in 5 of 10 cases, providing compelling evidence regarding the pathophysiology of NL. The conus medullaris was not radiologically involved in any case. Lumbosacral plexus MRI was able to identify extraspinal disease and offered diagnostically useful biopsy targets. FDG PET/CT was relatively insensitive for detecting disease in the cauda equina but was helpful in identifying extraspinal NL.

CONCLUSIONS

The authors propose that perineural spread of extraspinal NL to infiltrate the cauda equina occurs in two phases. 1) There is proximal and distal spread along a peripheral nerve, with eventual spread to anatomically connected nerves via junction and branch points. 2) The tumor cells enter the spinal canal through corresponding neural foramina and propagate along the spinal nerves composing the cauda equina. To diffusely infiltrate the cauda equina, a third phase occurs in which tumor cells can spread circumdurally to the opposite side of the spinal canal and enter contralateral nerve roots extending proximally and distally. This spread of disease can lead to diffuse bilateral spinal nerve disease without diffuse leptomeningeal spread. Recognition of this phasic mechanism can lead to identification of safer extraspinal biopsy targets that could allow for greater functional recovery after appropriate treatment.

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Mohammed Ali Alvi, Waseem Wahood, Shyam J. Kurian, Jad Zreik, Molly M. Jeffery, James M. Naessens, Robert J. Spinner, and Mohamad Bydon

OBJECTIVE

Spinal procedures are increasingly conducted as outpatient procedures, with a growing proportion conducted in ambulatory surgery centers (ASCs). To date, studies reporting outcomes and cost analyses for outpatient spinal procedures in the US have not distinguished the various outpatient settings from each other. In this study, the authors used a state-level administrative database to compare rates of overnight stays and nonroutine discharges as well as index admission charges and cumulative 7-, 30-, and 90-day charges for patients undergoing outpatient lumbar decompression in freestanding ASCs and hospital outpatient (HO) settings.

METHODS

For this project, the authors used the Florida State Ambulatory Surgery Database (SASD), offered by the Healthcare Cost and Utilization Project (HCUP), for the years 2013 and 2014. Patients undergoing outpatient lumbar decompression for degenerative diseases were identified using CPT (Current Procedural Terminology) and ICD-9 codes. Outcomes of interest included rates of overnight stays, rate of nonroutine discharges, index admission charges, and subsequent admission cumulative charges at 7, 30, and 90 days. Multivariable analysis was performed to assess the impact of outpatient type on index admission charges. Marginal effect analysis was employed to study the difference in predicted dollar margins between ASCs and HOs for each insurance type.

RESULTS

A total of 25,486 patients were identified; of these, 7067 patients (27.7%) underwent lumbar decompression in a freestanding ASC and 18,419 (72.3%) in an HO. No patient in the ASC group required an overnight stay compared to 9.2% (n = 1691) in the HO group (p < 0.001). No clinically significant difference in the rate of nonroutine discharge was observed between the two groups. The mean index admission charge for the ASC group was found to be significantly higher than that for the HO group ($35,017.28 ± $14,335.60 vs $33,881.50 ± $15,023.70; p < 0.001). Patients in ASCs were also found to have higher mean 7-day (p < 0.001), 30-day (p < 0.001), and 90-day (p = 0.001) readmission charges. ASC procedures were associated with increased charges compared to HO procedures for patients on Medicare or Medicaid (mean index admission charge increase $4049.27, 95% CI $2577.87–$5520.67, p < 0.001) and for patients on private insurance ($4775.72, 95% CI $4171.06–$5380.38, p < 0.001). For patients on self-pay or no charge, a lumbar decompression procedure at an ASC was associated with a decrease in index admission charge of −$10,995.38 (95% CI −$12124.76 to −$9866.01, p < 0.001) compared to a lumbar decompression procedure at an HO.

CONCLUSIONS

These “real-world” results from an all-payer statewide database indicate that for outpatient spine surgery, ASCs may be associated with higher index admission and subsequent 7-, 30-, and 90-day charges. Given that ASCs are touted to have lower overall costs for patients and better profit margins for physicians, these analyses warrant further investigation into whether this cost benefit is applicable to outpatient spine procedures.

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Anshit Goyal, Stephanie Payne, Lindsey R. Sangaralingham, Molly Moore Jeffery, James M. Naessens, Halena M. Gazelka, Elizabeth B. Habermann, William Krauss, Robert J. Spinner, and Mohamad Bydon

OBJECTIVE

Sustained postoperative opioid use after elective surgery is a matter of growing concern. Herein, the authors investigated incidence and predictors of long-term opioid use among patients undergoing elective lumbar spine surgery, especially as a function of opioid prescribing practices at postoperative discharge (dose in morphine milligram equivalents [MMEs] and type of opioid).

METHODS

The OptumLabs Data Warehouse (OLDW) was queried for postdischarge opioid prescriptions for patients undergoing elective lumbar decompression and discectomy (LDD) or posterior lumbar fusion (PLF) for degenerative spine disease. Only patients who received an opioid prescription at postoperative discharge and those who had a minimum of 180 days of insurance coverage prior to surgery and 180 days after surgery were included. Opioid-naive patients were defined as those who had no opioid fills in 180 days prior to surgery. The following patterns of long-term postoperative use were investigated: additional fills (at least one opioid fill 90–180 days after surgery), persistent fills (any span of opioid use starting in the 180 days after surgery and lasting at least 90 days), and Consortium to Study Opioid Risks and Trends (CONSORT) criteria for persistent use (episodes of opioid prescribing lasting longer than 90 days and 120 or more total days’ supply or 10 or more prescriptions in 180 days after the index fill). Multivariable logistic regression was performed to identify predictors of long-term use.

RESULTS

A total of 25,587 patients were included, of whom 52.7% underwent PLF (n = 13,486) and 32.5% (n = 8312) were opioid-naive prior to surgery. The rates of additional fills, persistent fills, and CONSORT use were 47%, 30%, and 23%, respectively, after PLF and 35.4%, 19%, and 14.2%, respectively, after LDD. The rates among opioid-naive patients were 18.9%, 5.6%, and 2.5% respectively, after PLF and 13.3%, 2.0%, and 0.8%, respectively, after LDD. Using multivariable logistic regression, the following were identified to be significantly associated with higher risk of long-term opioid use following PLF: discharge opioid prescription ≥ 500 MMEs, prescription of a long-acting opioid, female sex, multilevel surgery, and comorbidities such as depression and drug abuse (all p < 0.05). Elderly (age ≥ 65 years) and opioid-naive patients were found to be at lower risk (all p < 0.05). Similar results were obtained on analysis for LDD with the following significant additional risk factors identified: discharge opioid prescription ≥ 400 MMEs, prescription of tramadol alone at discharge, and inpatient surgery (all p < 0.05).

CONCLUSIONS

In an analysis of pharmacy claims from a national insurance database, the authors identified incidence and predictors of long-term opioid use after elective lumbar spine surgery.

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Anthony L. Mikula, Brandon W. Smith, Nikita Lakomkin, Matthew K. Doan, Megan M. Jack, Mohamad Bydon, and Robert J. Spinner

OBJECTIVE

The objective of this study was to determine if patients with nerve sheath tumors affecting the C5 spinal nerve are at greater risk for postoperative weakness than those with similar tumors affecting other spinal nerves contributing to the brachial plexus.

METHODS

A retrospective chart review (1998–2020)identified patients with pathologically confirmed schwannomas or neurofibromas from the C5 to T1 nerves. Patients with plexiform nerve sheath tumors, tumors involving more than 1 nerve, and malignant peripheral nerve sheath tumors were excluded. Collected variables included basic demographics, tumor dimensions, its location relative to the dura, involved nerve level, surgical approach, extent of resection, presenting symptoms, postoperative neurological deficits, and recurrence rate.

RESULTS

Forty-six patients (23 men, 23 women) were identified for inclusion in the study with an average age of 47 ± 17 years, BMI of 28 ± 5 kg/m, and follow-up of 32 ± 45 months. Thirty-nine patients (85%) had schwannomas and 7 (15%) had neurofibromas. Tumors involved the C5 (n = 12), C6 (n = 11), C7 (n = 14), C8 (n = 6), and T1 (n = 3) nerves. Multivariable logistic regression analysis with an area under the curve of 0.85 demonstrated C5 tumor level as an independent predictor of new postoperative weakness (odds ratio 7.4, p = 0.028). Of those patients with new postoperative weakness, 75% improved and 50% experienced complete resolution of their motor deficits.

CONCLUSIONS

Patients with C5 nerve sheath tumor resections are at higher odds of new postoperative weakness. This may be due to the predominant single innervation of shoulder muscle targets in contrast to other upper extremity muscles that receive input from 2 or more spinal nerves. These findings are important for clinical decision-making and preoperative patient counseling.

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Brandon W. Smith, Megan M. Jack, Garret M. Powell, Matthew A. Frick, Kimberly K. Amrami, and Robert J. Spinner

BACKGROUND

The advancement of high-resolution imaging and increased clinical experience have led to an increased understanding of the formation and treatment of intraneural ganglion cysts. Nearly all intraneural ganglion cysts in the common peroneal nerve have been reported to arise from a joint connection to the superior tibiofibular joint. The authors have identified four cases of intraneural ganglion cysts arising from the knee joint itself; however, none of these reported cases were well described, documented, or illustrated with high-resolution imaging.

OBSERVATIONS

Here the authors present the case of an intraneural ganglion cyst arising from the knee joint and causing intermittent weakness and pain. The articular branch to the knee joint was clearly demonstrated on high-resolution magnetic resonance imaging and confirmed at surgical exploration. The patient was treated with articular branch ligation and has had complete resolution of his symptoms without recurrence of the cyst on follow-up imaging.

LESSONS

This case adds to the mounting evidence that intraneural cyst pathology is dependent on a connection to a synovial joint as stated in the unifying theory of intraneural cyst development.

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Thomas J. Wilson, Forrest Hamrick, Saud Alzahrani, Christopher F. Dibble, Sravanthi Koduri, Courtney Pendleton, Sara Saleh, Zarina S. Ali, Mark A. Mahan, Rajiv Midha, Wilson Z. Ray, Lynda J. S. Yang, Eric L. Zager, and Robert J. Spinner

OBJECTIVE

The aim of this study was to examine the role of intraoperative neuromonitoring (IONM) during resection of benign peripheral nerve sheath tumors in achieving gross-total resection (GTR) and in reducing postoperative neurological complications.

METHODS

Data from consecutive adult patients who underwent resection of a benign peripheral nerve sheath tumor at 7 participating institutions were combined. Propensity score matching was used to balance covariates. The primary outcomes of interest were the association between IONM and GTR and the association of IONM and the development of a permanent postoperative neurological complication. The secondary outcomes of interest were the association between IONM and GTR and the association between IONM and the development of a permanent postoperative neurological complication in the subgroup of patients with tumors involving a motor or mixed nerve. Univariate and multivariate logistic regression were then performed on the propensity score–matched samples to assess the ability of the independent variables to predict the outcomes of interest.

RESULTS

A total of 337 patients who underwent resection of benign nerve sheath tumors were included. In multivariate analysis, the use of IONM (OR 0.460, 95% CI 0.199–0.978; p = 0.047) was a significant negative predictor of GTR, whereas none of the variables, including IONM, were associated with the occurrence of a permanent postoperative neurological complication. Within the subgroup of motor/mixed nerve tumors, in the multivariate analysis, IONM (OR 0.263, 95% CI 0.096–0.723; p = 0.010) was a significant negative predictor of a GTR, whereas IONM (OR 3.800, 95% CI 1.925–7.502; p < 0.001) was a significant positive predictor of a permanent postoperative motor deficit.

CONCLUSIONS

Overall, 12% of the cohort had a permanent neurological complication, with new or worsened paresthesias most common, followed by pain and then weakness. The authors found that formal IONM was associated with a reduced likelihood of GTR and had no association with neurological complications. The authors believe that these data argue against IONM being considered standard of care but do not believe that these data should be used to universally argue against IONM during resection of benign nerve sheath tumors.

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Victor M. Lu, S. Shelby Burks, Rainya N. Heath, Tizeta Wolde, Robert J. Spinner, and Allan D. Levi

OBJECTIVE

Meralgia paresthetica is caused by entrapment of the lateral femoral cutaneous nerve (LFCN) and often presents with pain. Multiple treatment options targeting the LFCN can be pursued to treat the pain should conservative measures fail, with the most common options being injection, neurolysis, and neurectomy. However, their efficacy in causing pain relief and their clinical outcomes have yet to be directly compared. The aim of this study was to interrogate the contemporary literature and quantitatively define how these options compare.

METHODS

The electronic databases Ovid Embase, PubMed, SCOPUS, and the Cochrane Library were interrogated from inception to May 2020 following the PRISMA guidelines. Candidate articles were screened against prespecified criteria. Outcome data were abstracted and pooled by random-effects meta-analysis of proportions.

RESULTS

There were 25 articles that satisfied all criteria, reporting outcomes for a total of 670 meralgia paresthetica patients, with 78 (12%) treated by injection, 496 (74%) by neurolysis, and 96 (14%) by neurectomy. The incidence of complete pain relief was 85% (95% CI 71%–96%) after neurectomy, 63% (95% CI 56%–71%) after neurolysis, and 22% (95% CI 13%–33%) after injection, which were all statistically different (p < 0.01). The incidence of revision procedures was 12% (95% CI 4%–22%) after neurolysis and 0% (95% CI 0%–2%) after neurectomy, which were significantly lower than 81% (95% CI 64%–94%) after injection (p < 0.01). The incidences of treatment complications were statistically comparable across all three treatments, ranging from 0% to 5% (p = 0.34).

CONCLUSIONS

There are multiple treatment options to target pain in meralgia paresthetica. The incidence of complete pain relief appears to be the greatest among the 3 interventions after neurectomy, accompanied by the lowest incidence of revision procedures. These findings should help inform patient preference and expectations. Greater exploration of the anatomical rationale for incomplete pain relief after surgical intervention will assist in optimizing further surgical treatment for meralgia paresthetica.

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Thomas J. Wilson, Andres A. Maldonado, Kimberly K. Amrami, Katrina N. Glazebrook, Michael R. Moynagh, and Robert J. Spinner

The authors present the cases of 3 patients with severe injuries affecting the peroneal nerve combined with loss of tibialis posterior function (inversion) despite preservation of other tibial nerve function. Loss of tibialis posterior function is problematic, since transfer of the tibialis posterior tendon is arguably the best reconstructive option for foot drop, when available. Analysis of preoperative imaging studies correlated with operative findings and showed that the injuries, while predominantly to the common peroneal nerve, also affected the lateral portion of the tibial nerve/division near the sciatic nerve bifurcation. Sunderland’s fascicular topographic maps demonstrate the localization of the fascicular bundle subserving the tibialis posterior to the area that corresponds to the injury. This has clinical significance in predicting injury patterns and potentially for treatment of these injuries. The lateral fibers of the tibial division/nerve may be vulnerable with long stretch injuries. Due to the importance of tibialis posterior function, it may be important to perform internal neurolysis of the tibial division/nerve in order to facilitate nerve action potential testing of these fascicles, ultimately performing split nerve graft repair when nerve action potentials are absent in this important portion of the tibial nerve.

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Hannah E. Gilder, Ross C. Puffer, Mohamad Bydon, and Robert J. Spinner

OBJECTIVE

In this study, the authors sought to compare tumors with intradural extension to those remaining in the epidural or paraspinal space with the hypothesis that intradural extension may be a mechanism for seeding of the CSF with malignant cells, thereby resulting in higher rates of CNS metastases and shorter overall survival.

METHODS

The authors searched the medical record for cases of malignant peripheral nerve sheath tumors (MPNSTs) identified from 1994 to 2017. The charts of the identified patients were then reviewed for tumor location to identify patients with paraspinal malignancy. All patients included in the study had tumor specimens that were reviewed in the surgical pathology department. Paraspinal tumors with intradural extension were identified in the lumbar, sacral, and spinal accessory nerves, and attempts were made to match this cohort to another cohort of patients who had paraspinal tumors of the cranial nerves and lumbar and sacral spinal regions without intradural extension. Further information was collected on all patients with and without intradural extension, including date of diagnosis by pathology specimen review; nerve or nerves of tumor origin; presence, location, and diagnostic date of any CNS metastases; and either the date of death or date of last follow-up.

RESULTS

The authors identified 6 of 179 (3.4%) patients who had intradural tumor extension and compared these patients with 12 patients who harbored paraspinal tumors that did not have intradural extension. All tumors were diagnosed as high-grade MPNSTs according to the surgical pathology findings. Four of 6 (66.7%) patients with intradural extension had documented CNS metastases. The presence of CNS metastases was significantly higher in the intradural group than in the paraspinal group (intradural, 66.7% vs paraspinal, 0%; p < 0.01). Time from diagnosis until death was 11.2 months in the intradural group and approximately 72 months in the paraspinal, extradural cohort.

CONCLUSIONS

In patients with intradural extension of paraspinal MPNSTs, significantly higher rates of CNS metastases are seen with a reduced interval of time from diagnosis to metastatic lesion detection. Intradural tumor extension is also a poor prognostic factor for survival, with these patients showing a reduced mean time from diagnosis to death.