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Letter to the Editor

Hip- and pelvic-related intraneural ganglia

Robert J. Spinner, Marie-Noëlle Hébert-Blouin, Shota Tanaka, Kimberly K. Amrami, Karin R. Swartz, Dominic B. Fee, and Makoto Sugita

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Multinodular/plexiform (multifascicular) schwannomas of major peripheral nerves: an underrecognized part of the spectrum of schwannomas

Clinical article

Marie-Noëlle Hébert-Blouin, Kimberly K. Amrami, Bernd W. Scheithauer, and Robert J. Spinner

Object

In clinical practice, schwannomas are among the most common types of nerve sheath tumors. Their clinical presentation, imaging characteristics, and operative features are well known. Over the past 20 years, clinical outcomes have improved due to resection of these tumors at a fascicular level. Despite these advances, a subgroup of patients with schwannomas is associated with a disappointing neurological outcome following resection. The purpose of this study was to correlate the imaging and histological features in this group of patients with more anatomically complex forms of schwannomas.

Methods

In a retrospective review performed at their institution over a 10-year period, the authors found a subgroup of patients with complex multinodular/plexiform schwannomas affecting major peripheral nerves. Eleven patients were identified, and the clinical, imaging, and pathological features of their disease were reviewed.

Results

The clinical presentation of multinodular/plexiform schwannomas of major peripheral nerves may be similar to that of conventional schwannomas, but their imaging features, operative appearance, and outcomes differ.

Conclusions

Preoperatively and intraoperatively, the distinguishing features of multinodular/plexiform schwannomas of major peripheral nerves may be subtle and can easily go unrecognized, thus explaining the often suboptimal surgical results. Familiarity with the imaging and operative features of multinodular/plexiform schwannomas will no doubt alter treatment approaches and improve neurological function in this subgroup of patients.

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Evidence that atypical juxtafacet cysts are joint derived

Clinical article

Robert J. Spinner, Marie-Noëlle Hébert-Blouin, Timothy P. Maus, John L. D. Atkinson, Nicholas M. Desy, and Kimberly K. Amrami

Object

Juxtafacet cysts (JFCs) in usual locations have recently been shown to have joint connections. The pathogenesis of JFCs in unusual locations has remained obscure. The authors hypothesize that all JFCs, including atypical ones, are joint derived.

Methods

In this study the authors sought to explain the occurrence and formation of clinical outliers of spinal JFCs. In Part I, they performed an extensive literature search to identify case reports of spinal intraneural cysts that have been unappreciated despite the fact that they should occur. In Part II, they studied far-lateral (extraforaminal) cysts treated at their institution and reported in the literature. The presence of a joint connection was specifically looked for since this finding has not been widely appreciated.

Results

In Part I, 3 isolated case reports of spinal intraneural JFCs without reported joint connections were identified: 2 involving L-5 and 1, C-8. In Part II, 6 cases involving patients with far-lateral JFCs treated at the authors' institution were reviewed and all 6 had joint connections. Two of these cases had been previously published, although their joint connections were not appreciated. In 2 of the newly reported cases, arthrography confirmed a communication between the facet and the cyst. Only 1 of 5 cases in the literature had a recognized joint connection.

Conclusions

The authors believe that all JFCs are joint derived. This explanation for intraneural and extraneural JFCs in typical locations would be consistent with the unified articular (synovial) theory and the pathogenesis for intraneural and extraneural ganglion cyst formation in the limbs. Facet joints appear no different from other synovial joints occurring elsewhere. Understanding the pathogenesis of these cysts will help target treatment to the joint, improve surgical outcomes, and decrease recurrences.

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Importance of the vasculature in cyst formation after spinal cord injury

Laboratory investigation

Gemma E. Rooney, Toshiki Endo, Syed Ameenuddin, Bingkun Chen, Sandeep Vaishya, LouAnn Gross, Terry K. Schiefer, Bradford L. Currier, Robert J. Spinner, Michael J. Yaszemski, and Anthony J. Windebank

Object

Glial scar and cystic formation greatly contribute to the inhibition of axonal regeneration after spinal cord injury (SCI). Attempts to promote axonal regeneration are extremely challenging in this type of hostile environment. The objective of this study was to examine the surgical methods that may be used to assess the factors that influence the level of scar and cystic formation in SCI.

Methods

In the first part of this study, a complete transection was performed at vertebral level T9–10 in adult female Sprague-Dawley rats. The dura mater was either left open (control group) or was closed using sutures or hyaluronic acid. In the second part of the study, complete or subpial transection was performed, with the same dural closure technique applied to both groups. Histological analysis of longitudinal sections of the spinal cord was performed, and the percentage of scar and cyst formation was determined.

Results

Dural closure using sutures resulted in significantly less glial scar formation (p = 0.0248), while incorporation of the subpial transection surgical technique was then shown to significantly decrease cyst formation (p < 0.0001).

Conclusions

In this study, the authors demonstrated the importance of the vasculature in cyst formation after spinal cord trauma and confirmed the importance of dural closure in reducing glial scar formation.

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Intraneural ganglia in the hip and pelvic region

Clinical article

Robert J. Spinner, Marie-Noëlle Hébert-Blouin, Robert T. Trousdale, Rajiv Midha, Stephen M. Russell, Tatsuya Yamauchi, Syouzou Sasaki, and Kimberly K. Amrami

Object

The authors describe their experience in a series of cases of intraneural ganglia within the hip and pelvic regions, and explain the mechanism of formation and propagation of this pathological entity.

Methods

Five patients with 6 intraneural ganglia are presented. Four patients presented with symptomatic intraneural ganglia in the buttock and pelvis affecting the sciatic and lumbosacral plexus elements. An asymptomatic cyst affecting the opposite sciatic nerve was found on MR imaging in 1 patient. The fifth patient, previously reported on by another group, had an obturator intraneural ganglion that the authors reinterpreted.

Results

All 5 intraneural ganglia affecting the sciatic and lumbosacral plexus elements were found to have a joint connection to the posteromedial aspect of the hip joint; the obturator intraneural cyst had a joint connection to the anteromedial aspect of the hip joint. In all cases, initial review of the MR images led to their misinterpretation.

Conclusions

To the authors' knowledge, these are the first cases of intraneural ganglia demonstrated to have a connection to the hip joint. This finding at a rare site provides further evidence for the unifying articular (synovial) theory for the formation of intraneural ganglia and reveals a shared mechanism for their propagation. Furthermore, understanding the pathogenesis of these lesions provides insight into their successful treatment and their recurrence.

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Adherence of intraneural ganglia of the upper extremity to the principles of the unifying articular (synovial) theory

Huan Wang, Robert Q. Terrill, Shota Tanaka, Kimberly K. Amrami, and Robert J. Spinner

Object

Intraneural ganglia are nonneoplastic mucinous cysts contained within the epineurium of peripheral nerves. Their pathogenesis has been controversial. Historically, the majority of authors have favored de novo formation (degenerative theory). Because of their rarity, intraneural ganglia affecting the upper limb have been misunderstood. This study was designed to critically analyze the literature and to test the hypothesis that intraneural ganglia of the upper limb act analogously to those in the lower limb, being derived from an articular source (synovial theory).

Methods

Two patients with digital intraneural cysts were included in the study. An extensive literature review of intraneural ganglia of the upper limb was undertaken to provide the historical basis for the study.

Results

In both cases, the digital intraneural ganglia were demonstrated to have joint connections; the one patient in whom an articular branch was not appreciated initially had evidence on postoperative MR images of persistence of intraneural cyst after simple decompression was performed. Eighty-six cases of intraneural lesions were identified in varied locations of the upper limb: the most common sites were the ulnar nerve at the elbow and wrist, occurring 38 and 22 times, respectively. Joint connections were present in only 20% of the cases published by other groups.

Conclusions

The authors believe that the fundamental principles of the unifying articular (synovial) theory (that is, articular branch connections, cyst fluid following a path of least resistance, and the role of pressure fluxes) previously described to explain intraneural ganglia in the lower limb apply to those cases in the upper limb. In their opinion, the joint connection is often not identified because of the cysts' rarity, radiologists' and surgeons' inexperience, and the difficulty visualizing and demonstrating it because of the small size of the cysts. Furthermore, they believe that recurrence (subclinical or clinical) is not only underreported but also predictable after simple decompression that fails to address the articular branch. In contrast, intraneural recurrence can be eliminated with disconnection of the articular branch.

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Designing ideal conduits for peripheral nerve repair

Godard C. W. de Ruiter, Martijn J. A. Malessy, Michael J. Yaszemski, Anthony J. Windebank, and Robert J. Spinner

Nerve tubes, guides, or conduits are a promising alternative for autologous nerve graft repair. The first biodegradable empty single lumen or hollow nerve tubes are currently available for clinical use and are being used mostly in the repair of small-diameter nerves with nerve defects of < 3 cm. These nerve tubes are made of different biomaterials using various fabrication techniques. As a result these tubes also differ in physical properties. In addition, several modifications to the common hollow nerve tube (for example, the addition of Schwann cells, growth factors, and internal frameworks) are being investigated that may increase the gap that can be bridged. This combination of chemical, physical, and biological factors has made the design of a nerve conduit into a complex process that demands close collaboration of bioengineers, neuroscientists, and peripheral nerve surgeons. In this article the authors discuss the different steps that are involved in the process of the design of an ideal nerve conduit for peripheral nerve repair.

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Intraneural ganglia: a clinical problem deserving a mechanistic explanation and model

Shreehari Elangovan, Gregory M. Odegard, Duane A. Morrow, Huan Wang, Marie-Noëlle Hébert-Blouin, and Robert J. Spinner

Intraneural ganglion cysts have been considered a curiosity for 2 centuries. Based on a unifying articular (synovial) theory, recent evidence has provided a logical explanation for their formation and propagation. The fundamental principle is that of a joint origin and a capsular defect through which synovial fluid escapes following the articular branch, typically into the parent nerve. A stereotypical, reproducible appearance has been characterized that suggests a shared pathogenesis. In the present report the authors will provide a mechanistic explanation that can then be mathematically tested using a preliminary model created by finite element analysis.

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Quantitative evaluation of movement and strength of the upper limb after transection of the C-7 nerve: is it possible in an animal model?

Laboratory investigation

Huan Wang, Robert J. Spinner, and Anthony J. Windebank

Object

Contralateral C-7 nerve transfer has been used clinically for more than 20 years. The increased interest in studies of transfer effectiveness at different target muscles, posttransfer cocontraction, and brain plasticity has prompted the need for an animal model. In addition to the conventional electrophysiological, histomorphometric, and biomechanical evaluation modalities, quantitative functional and behavioral evaluation will be crucial in applying this kind of model. The aim of this study was to establish a C-7 transection animal model and quantify the changes in upper-limb joint movement and muscle power.

Methods

A C-7 nerve transection model was created in Sprague-Dawley rats, the brachial plexus of which resembles the human brachial plexus. The impact of C-7 transection on donor limb function—namely, strength, movement, and coordination—was evaluated in 6 rats. Muscle strength (power reported in g) was measured as a grasping task. The active range of motion (ROM; angle reported in °) of the elbow, wrist, and metacarpophalangeal joints was quantified by computerized video motion analysis. Antiresistance coordinated movement (speed reported in seconds) was assessed by the vertical rope-climbing test. These tests were carried out before surgery and at 2, 4, 6, 8, 10, 14, 21, and 28 days after C-7 transection. Repeated-measures 1-way analysis of variance was applied for statistical analysis. When the overall probability value was < 0.05, the Dunnett multiple-comparison posttest was used to compare postoperative values with preoperative baseline values.

Results

Immediately after C-7 transection, the mean ± SD grip strength declined from 378.50 ± 20.55 g to 297.77 ± 15.04 g. Active elbow extension was impaired, as shown by a significant decrease of the elbow extension angle. The speed of vertical rope climbing was also reduced. Elbow flexion, wrist flexion and extension, and metacarpophalangeal joint flexion and extension were not impaired. Fast recovery of motor function was observed thereafter. Grip strength, range of active elbow extension, and speed of rope climbing returned to baseline values at postoperative Days 4, 8, and 8, respectively.

Conclusions

The ROM and muscle strength of the upper limb in rats can be measured quantitatively in studies that simulate clinical situations. Application of these functional evaluation modalities in a C-7 nerve transection rat model confirmed that transection of C-7 causes only temporary functional dysfunction to the donor limb. The results obtained in this animal model mimic those seen in humans who undergo contralateral C-7 nerve harvesting.

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Glomus tumor and glomangioma of the nerve

Report of 2 cases

Bernd W. Scheithauer, Fausto J. Rodriguez, Robert J. Spinner, P. James Dyck, Ayman Salem, Fredric L. Edelman, Kimberly K. Amrami, and Yao-Shi Fu

✓ As a rule, normal human nerve does not contain glomus bodies. Nonetheless, rare examples of glomus tumors do arise in peripheral nerves of various sizes. Their pathobiological characteristics are poorly understood, but reported examples have been small and clinically benign. The authors identified in 1 patient each a glomus tumor and a glomangioma involving nerve. Clinical histories as well as imaging and surgical findings were reviewed. All available H & E–stained slides were examined in both cases. Immunohistochemical stains and electron microscopy, as appropriate, were also performed. The lesions were subtotally and completely resected, respectively. An uneventful postoperative recovery was noted in both patients. Glomus tumors and glomangiomas can involve major nerves on rare occasions. They seem to follow a favorable clinical course, and conservative resection can be of benefit.