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Mohammed Adeeb Sebai, Panagiotis Kerezoudis, Mohammed Ali Alvi, Jang Won Yoon, Robert J. Spinner and Mohamad Bydon

OBJECTIVE

Spinal peripheral nerve sheath tumors (PNSTs) are a group of rare tumors originating from the nerve and its supporting structures. Standard surgical management typically entails laminectomy with or without facetectomy to gain adequate tumor exposure. Arthrodesis is occasionally performed to maintain spinal stability and mitigate the risk of postoperative deformity, pain, or neurological deficit. However, the factors associated with the need for instrumentation in addition to PNST resection in the same setting remain unclear.

METHODS

An institutional tumor registry at a tertiary care center was queried for patients treated surgically for a primary diagnosis of spinal PNST between 2002 and 2016. An analysis focused on patients in whom a facetectomy was performed during the resection. The addition of arthrodesis at the index procedure comprised the primary outcome. The authors also recorded baseline demographics, tumor characteristics, and surgery-related variables. Logistic regression was used to identify factors associated with increased risk of fusion surgery.

RESULTS

A total of 163 patients were identified, of which 56 (32 had facetectomy with fusion, 24 had facetectomy alone) were analyzed. The median age was 48 years, and 50% of the cohort was female. Age, sex, and race, as well as tumor histology and size, were evenly distributed between patients who received facetectomy alone and those who had facetectomy and fusion. On univariate analysis, total versus subtotal facetectomy (OR 9.0, 95% CI 2.01–64.2; p = 0.009) and cervicothoracic versus other spinal region (OR 9.0, 95% CI 1.51–172.9; p = 0.048) were significantly associated with increased odds of performing immediate fusion. On multivariable analysis, only the effect of total facetectomy remained statistically significant (OR 6.75, 95% CI 1.47–48.8; p = 0.025).

CONCLUSIONS

The authors found that total facetectomy and cervicothoracic involvement may be highly associated with the need for concomitant arthrodesis at the time of index surgery. These findings may help surgeons to determine the best surgical planning for patients with PNST.

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Tomas Marek, Robert J. Spinner, Akshay Syal and Mark A. Mahan

OBJECTIVE

Lipomatosis of nerve (LN) is a massive enlargement of a nerve due to abundant proliferation of adipose and fibrotic tissue within the epineurium—part of the spectrum of adipose lesions of nerves, including intra- and extraneural lipomas. LN has been frequently associated with soft-tissue and/or osseous overgrowth. Unfortunately, much confusion exists since many names have been used for LN (e.g., fibrolipomatous hamartoma, macrodystrophia lipomatosa, and so on). To better understand this condition and to evaluate its association with nerve-territory overgrowth, the authors attempted to compile the world’s literature on published LN cases.

METHODS

PubMed and Google Scholar databases were searched to identify published articles on LN cases, using a variety of terms. Publications in all languages were assessed. All publications with cases determined likely to be LN were read. Cases that provided clear clinicopathological and/or radiological evidence of LN were labeled as “definite” and cases that demonstrated features of LN (e.g., nerve-territory overgrowth) but lacked definite proof of nerve involvement were labeled as “probable.”

RESULTS

Initial screening revealed a total of 2465 papers. After exclusions, 281 publications reported cases with a definite diagnosis of LN and 120 articles reported cases with a probable diagnosis of LN. The authors identified 618 definite and 407 probable cases of LN. Sex distribution was balanced (51% female). Early diagnosis was common, with two-thirds of patients having symptoms in the 1st decade of life. The most commonly affected nerve was the median nerve (n = 391). Nerve-territory overgrowth was common (62% definite LN; 78% combined cases); overgrowth was exclusive to the territory of the affected nerve in all cases but 5.

CONCLUSIONS

The authors present a comprehensive review and analysis of the literature of LN cases. One of the main findings was the nerve-territory overgrowth was associated with LN, especially when present earlier in life. The authors believe that all cases of LN associated with overgrowth can be explained on anatomical grounds, even in the few reported cases in which this is not immediately obvious.

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Aaron P. Kamer, Jose M. Bonnin, Robert J. Spinner and Aaron A. Cohen-Gadol

Intracranial extension of temporomandibular joint (TMJ) ganglion cysts is very rare. Two previously reported cases presented clinically due to effects on cranial nerves and had obvious association with the TMJ on imaging. To the authors’ knowledge, intracranial extension of a TMJ ganglion cyst presenting with seizures and mimicking a primary brain tumor has not been previously reported. The patient underwent resection of a presumptive primary cystic temporal lobe tumor, but the lesion had histopathological features of a nonneoplastic cyst with a myxoid content. He was followed with serial imaging for 5 years before regrowth of the lesion caused new episodes of seizures requiring a repeat operation, during which the transdural defect was repaired after the adjacent segment of the TMJ was curetted. A thorough review of all imaging studies and the histopathological findings from the repeat operation led to the correct diagnosis of a TMJ ganglion cyst. This case highlights an unusual presentation of this rare lesion, as well as its potential for recurrence. TMJ ganglion cysts should be included in the differential diagnosis of cystic tumors involving the anterior temporal lobe, presenting with or without seizures. Focused imaging evaluation of the TMJ can be helpful to rule out the possible role of associated TMJ lesions.

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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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Thomas J. Wilson, B. Matthew Howe, Shelby A. Stewart, Robert J. Spinner and Kimberly K. Amrami

OBJECTIVE

This study aimed to define a set of clinicoradiological parameters with a high specificity for the diagnosis of intraneural perineurioma, obviating the need for operative tissue diagnosis.

METHODS

The authors retrospectively reviewed MR images obtained in a large cohort of patients who underwent targeted fascicular biopsy and included only those patients for whom the biopsy yielded a diagnosis. Clinical and radiological findings were then tested for their ability to predict a tissue diagnosis of intraneural perineurioma. The authors propose a new set of diagnostic criteria, referred to as the Perineurioma Diagnostic Criteria. The sensitivity, specificity, positive predictive value, and negative predictive value of several clinicoradiological methods of diagnosis were compared.

RESULTS

A total of 195 patients who underwent targeted fascicular biopsy were included in the cohort, of whom 51 had a tissue diagnosis of intraneural perineurioma. When the clinicoradiological methods used in this study were compared, the highest sensitivity (0.86), negative predictive value (0.95), and F1 score (0.88) were observed for the decision trees generated in C5.0 and rPart, whereas the highest specificity (1.0) and positive predictive value (1.0) were observed for the Perineurioma Diagnostic Criteria.

CONCLUSIONS

This study identified clinical and radiological features that are associated with a diagnosis of perineurioma. The Perineurioma Diagnostic Criteria were determined to be the following: 1) no cancer history, 2) unifocal disease, 3) moderate to severe hyperintensity on T2-weighted MR images, 4) moderate to severe contrast enhancement, 5) homogeneous contrast enhancement, 6) fusiform shape, 7) enlargement of the involved nerves, and 8) age ≤ 40 years. Use of the Perineurioma Diagnostic Criteria obviates the need for tissue diagnosis when all of the criteria are satisfied.

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Liselotte F. Bulstra, Nadia Rbia, Michelle F. Kircher, Robert J. Spinner, Allen T. Bishop and Alexander Y. Shin

Despite continuous improvement and expansion of reconstructive options for traumatic brachial plexus injury, options to reinnervate the triceps muscle remain somewhat sparse. This study describes a novel option, using a spinal accessory nerve transfer to the long head of the triceps muscle with an intervening autologous nerve graft. The resulting quality of elbow extension and factors that influence outcome are discussed.

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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.

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Jonathan J. Stone, Nikhil K. Prasad, Pierre Laumonerie, B. Matthew Howe, Kimberly K. Amrami, Jodi M. Carter, Mark E. Jentoft and Robert J. Spinner

OBJECTIVE

Desmoid-type fibromatosis (DTF) presents a therapeutic dilemma. While lacking metastatic potential, it is a locally aggressive tumor with a strong propensity for occurrence near nerve(s) and recurrence following resection. In this study, the authors introduce the association of an occult neuromuscular choristoma (NMC) identified in patients with DTF.

METHODS

After experiencing a case of DTF found to have an occult NMC, the authors performed a retrospective database review of all other cases of biopsy-proven DTF involving the extremities or limb girdles in patients with available MRI data. Two musculoskeletal radiologists with expertise in peripheral nerve imaging reviewed the MRI studies of the eligible cases for evidence of previously unrecognized NMC.

RESULTS

The initial case of a patient with an occult sciatic NMC is described. The database review yielded 40 patients with DTF—18 (45%) in the upper limb and 22 (55%) in the lower limb. Two cases (5%) had MRI findings of NMC associated with the DTF, one in the proximal sciatic nerve and the other in the proximal tibial and sural nerves.

CONCLUSIONS

The coexistence of NMC may be under-recognized in a subset of patients with extremity DTF. This finding poses implications for DTF treatment and the likelihood of recurrence after resection or biopsy. Further study may reveal crucial links between the pathogenesis of NMC and DTF and offer novel therapeutic strategies.

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Benjamin T. Himes, Thomas J. Wilson, Andres A. Maldonado, Naveen S. Murthy and Robert J. Spinner

The authors present a case of delayed peroneal neuropathy following a lateral gastrocnemius rotational flap reconstruction. The patient presented 1.5 years after surgery with a new partial foot drop, which progressed over 3 years. At operation, a fascial band on the deep side of the gastrocnemius flap was compressing the common peroneal nerve proximal to the fibular head, correlating with preoperative imaging. Release of this fascial band and selective muscle resection led to immediate improvement in symptoms postoperatively.

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Courtney Pendleton, Allan J. Belzberg, Robert J. Spinner and Alfredo Quinones-Hinojosa

Harvey Cushing is widely regarded as one of the forefathers of neurosurgery, and is primarily associated with his work on intracranial pathology. However, he had a clinical and academic interest in peripheral nerve surgery. Through the courtesy of the Alan Mason Chesney Medical Archives, the surgical records of the Johns Hopkins Hospital from 1896 to 1912 were reviewed. The records of a single patient undergoing brachial plexus exploration and cervical rib resection were selected for detailed review. The operative report and accompanying illustrations demonstrate Cushing’s interest in adding approaches to the pathology of the brachial plexus to his operative armamentarium.