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Rinchen Phuntsok, Benjamin J. Ellis, Michael R. Herron, Chase W. Provost, Andrew T. Dailey and Douglas L. Brockmeyer

OBJECTIVE

There is contradictory evidence regarding the relative contribution of the key stabilizing ligaments of the occipitoatlantal (OA) joint. Cadaveric studies are limited by the nature and the number of injury scenarios that can be tested to identify OA stabilizing ligaments. Finite element (FE) analysis can overcome these limitations and provide valuable data in this area. The authors completed an FE analysis of 5 subject-specific craniocervical junction (CCJ) models to investigate the biomechanics of the OA joint and identify the ligamentous structures essential for stability.

METHODS

Isolated and combined injury scenarios were simulated under physiological loads for 5 validated CCJ FE models to assess the relative role of key ligamentous structures on OA joint stability. Each model was tested in flexion-extension, axial rotation, and lateral bending in various injury scenarios. Isolated ligamentous injury scenarios consisted of either decreasing the stiffness of the OA capsular ligaments (OACLs) or completely removing the transverse ligament (TL), tectorial membrane (TM), or alar ligaments (ALs). Combination scenarios were also evaluated.

RESULTS

An isolated OACL injury resulted in the largest percentage increase in all ranges of motion (ROMs) at the OA joint compared with the other isolated injuries. Flexion, extension, lateral bending, and axial rotation significantly increased by 12.4% ± 7.4%, 11.1% ± 10.3%, 83.6% ± 14.4%, and 81.9% ± 9.4%, respectively (p ≤ 0.05 for all). Among combination injuries, OACL+TM+TL injury resulted in the most consistent significant increases in ROM for both the OA joint and the CCJ during all loading scenarios. OACL+AL injury caused the most significant percentage increase for OA joint axial rotation.

CONCLUSIONS

These results demonstrate that the OACLs are the key stabilizing ligamentous structures of the OA joint. Injury of these primary stabilizing ligaments is necessary to cause OA instability. Isolated injuries of TL, TM, or AL are unlikely to result in appreciable instability at the OA joint.

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Garrett K. Berger, Peyton L. Nisson, Whitney S. James, Kristen N. Kaiser and R. John Hurlbert

OBJECTIVE

Ewing sarcoma (ES) is among the most prevalent of bone sarcomas in young people. Less often, it presents as a primary lesion of the spine (5%–15% of patients with ES).

METHODS

A systematic literature search was performed, querying several scientific databases per PRISMA guidelines. Inclusion criteria specified all studies of patients with surgically treated ES located in the spine. Patient age was categorized into three groups: 0–13 years (age group 1), 14–20 years (age group 2), and > 21 (age group 3).

RESULTS

Eighteen studies were included, yielding 28 patients with ES of the spine. Sixty-seven percent of patients experienced a favorable outcome, with laminectomies representing the most common (46%) of surgical interventions. One-, 2-, and 5-year survival rates were 82% (n = 23), 75% (n = 21), and 57% (n = 16), respectively. Patients in age group 2 experienced the greatest mortality rate (75%) compared to age group 1 (9%) and age group 3 (22%). The calculated relative risk score indicated patients in age group 2 were 7.5 times more likely to die than other age groups combined (p = 0.02).

CONCLUSIONS

Primary ES of the spine is a rare, debilitating disease in which the role of surgery and its impact on one’s quality of life and independence status has not been well described. This study found the majority of patients experienced a favorable outcome with respect to independence status following surgery and adjunctive treatment. An increased risk of recurrence and death was also present among the adolescent age group (14–20 years).

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Yair M. Gozal, Gmaan Alzhrani, Hussam Abou-Al-Shaar, Mohammed A. Azab, Michael T. Walsh and William T. Couldwell

OBJECTIVE

Cavernous sinus meningiomas are complex tumors that offer a perpetual challenge to skull base surgeons. The senior author has employed a management strategy for these lesions aimed at maximizing tumor control while minimizing neurological morbidity. This approach emphasizes combining “safe” tumor resection and direct decompression of the roof and lateral wall of the cavernous sinus as well as the optic nerve. Here, the authors review their experience with the application of this technique for the management of cavernous sinus meningiomas over the past 15 years.

METHODS

A retrospective analysis was performed for patients with cavernous sinus meningiomas treated over a 15-year period (2002–2017) with this approach. Patient outcomes, including cranial nerve function, tumor control, and surgical complications were recorded.

RESULTS

The authors identified 50 patients who underwent subtotal resection via frontotemporal craniotomy concurrently with decompression of the cavernous sinus and ipsilateral optic nerve. Of these, 25 (50%) underwent adjuvant radiation to the remaining tumor within the cavernous sinus. Patients most commonly presented with a cranial nerve (CN) palsy involving CN III–VI (70%), a visual deficit (62%), headaches (52%), or proptosis (44%). Thirty-five patients had cranial nerve deficits preoperatively. In 52% of these cases, the neuropathy improved postoperatively; it remained stable in 46%; and it worsened in only 2%. Similarly, 97% of preoperative visual deficits either improved or were stable postoperatively. Notably, 12 new cranial nerve deficits occurred postoperatively in 10 patients. Of these, half were transient and ultimately resolved. Finally, radiographic recurrence was noted in 5 patients (10%), with a median time to recurrence of 4.6 years.

CONCLUSIONS

The treatment of cavernous sinus meningiomas using surgical decompression with or without adjuvant radiation is an effective oncological strategy, achieving excellent tumor control rates with low risk of neurological morbidity.

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Constanze Buus-Gehrig, Thomas Lehrnbecher, Luciana Porto, Martina Becker, Thomas Freiman, Michel Mittelbronn and Konrad Bochennek

Tumors of the central nervous system represent the largest group of solid tumors found in pediatric patients. Pilocytic astrocytoma is the most common pediatric glioma, mostly located in the posterior fossa. The majority of brainstem tumors, however, are classified as highly aggressive diffuse intrinsic pontine gliomas (DIPGs) and their prognosis is dismal.

The authors report on the case of a neonate in whom MRI and neuropathological assessment were used to diagnose DIPG. Before initiation of the planned chemotherapy, the tumor regressed spontaneously, and the newborn exhibited a normal neurological development. Meanwhile, Illumina Human Methylation450 BeadChip analysis reclassified the tumor as pilocytic astrocytoma of the posterior fossa.

In conclusion, the authors advocate not initiating immediate intensive therapy in newborns with brain tumors, even with classical appearance of a DIPG; rather, they would like to encourage a biopsy to define the best individual therapeutic approach and avoid ineffective chemotherapy.

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Richard Menger, Patrick Kelly, Shanik Fernando, Michael E. Wolf and Anthony Martino

On May 5, 1961, Alan B. Shepard Jr. piloted the Freedom 7 craft into a suborbital flight to become the first American man in space. His promising astronautical career was soon scuttled by spells of dizziness and tinnitus later diagnosed as Ménière’s disease, until William F. House—considered the father of neurotology and a pioneer in surgery for vestibular schwannomas—intervened. In 1968 House implanted an endolymphatic-subarachnoid shunt, which at the time was a virtually experimental procedure. Shepard’s debilitating Ménière’s disease was cured, but not quite in time for him to pilot the doomed Apollo 13 mission; he was reassigned to Apollo 14 and as a result would step foot on the moon on February 5, 1971. This historical vignette depicts the tale of how the career trajectories of Shepard and House—two notable figures in their respective fields—fatefully intersected.

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Pepijn O. Sun, Ruud W. Selles, Miguel C. Jansen, Harm P. Slijper, Dietmar J. O. Ulrich and Erik T. Walbeehm

OBJECTIVE

The aim of this study was to evaluate the self-reported outcome of revision surgery in patients with recurrent and persistent carpal tunnel syndrome (CTS) and to identify predictors of clinical outcome of revision surgery.

METHODS

A total of 114 hands in 112 patients were surgically treated for recurrent and persistent CTS in one of 10 specialized hand clinics. As part of routine care, patients were asked to complete online questionnaires regarding demographic data, comorbidities, and clinical severity measures. The Boston Carpal Tunnel Questionnaire (BCTQ) was administered at intake and at 6 months postoperatively to evaluate clinical outcome. The BCTQ comprises the subscales Symptom Severity Scale (SSS) and Functional Status Scale (FSS), and the individual scores were also assessed. Using multivariable regression models, the authors identified factors predictive of the outcome as measured by the BCTQ FSS, SSS, and total score at 6 months.

RESULTS

Revision surgery significantly improved symptoms and function. Longer total duration of symptoms, a higher BCTQ total score at intake, and diagnosis of complex regional pain syndrome (CRPS) along with CTS were associated with worse outcome after revision surgery at 6 months postoperatively. The multivariable prediction models could explain 33%, 23%, and 30% of the variance in outcome as measured by the FSS, SSS, and BCTQ total scores, respectively, at 6 months. Although patients with higher BCTQ scores at intake have worse outcomes, they generally have the most improvement in symptoms and function.

CONCLUSIONS

This study identified total duration of symptoms, BCTQ total score at intake, and diagnosis of CRPS along with CTS as predictors of clinical outcome and confirmed that revision surgery significantly improves self-reported symptoms and function in patients with recurrent and persistent CTS. Patients with more severe CTS symptoms have greater improvement in symptoms at 6 months postoperatively than patients with less severe CTS, but 80% of patients still had residual symptoms 6 months postoperatively. These results can be used to inform both patient and surgeon to manage expectations on improvement of symptoms.

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Tamara D. Simon, Matthew P. Kronman, Kathryn B. Whitlock, Samuel R. Browd, Richard Holubkov, John R. W. Kestle, Abhaya V. Kulkarni, Marcie Langley, David D. Limbrick Jr., Thomas G. Luerssen, W. Jerry Oakes, Jay Riva-Cambrin, Curtis Rozzelle, Chevis N. Shannon, Mandeep Tamber, John C. Wellons III, William E. Whitehead and Nicole Mayer-Hamblett

OBJECTIVE

CSF shunt infection treatment requires both surgical and antibiotic decisions. Using the Hydrocephalus Clinical Research Network (HCRN) Registry and 2004 Infectious Diseases Society of America (IDSA) guidelines that were not proactively distributed to HCRN providers, the authors previously found high adherence to surgical recommendations but poor adherence to intravenous (IV) antibiotic duration recommendations. In general, IV antibiotic duration was longer than recommended. In March 2017, new IDSA guidelines expanded upon the 2004 guidelines by including recommendations for selection of specific antibiotics. The objective of this study was to describe adherence to both 2004 and 2017 IDSA guideline recommendations for CSF shunt infection treatment, and to report reinfection rates associated with adherence to guideline recommendations.

METHODS

The authors investigated a prospective cohort of children younger than 18 years of age who underwent treatment for first CSF shunt infection at one of 7 hospitals from April 2008 to December 2012. CSF shunt infection was diagnosed by recovery of bacteria from CSF culture (CSF-positive infection). Adherence to 2004 and 2017 guideline recommendations was determined. Adherence to antibiotics was further classified as longer or shorter duration than guideline recommendations. Reinfection rates with 95% confidence intervals (CIs) were generated.

RESULTS

There were 133 children with CSF-positive infections addressed by 2004 IDSA guideline recommendations, with 124 at risk for reinfection. Zero reinfections were observed among those whose treatment was fully adherent (0/14, 0% [95% CI 0%–20%]), and 15 reinfections were observed among those whose infection treatment was nonadherent (15/110, 14% [95% CI 8%–21%]). Among the 110 first infections whose infection treatment was nonadherent, 74 first infections were treated for a longer duration than guidelines recommended and 9 developed reinfection (9/74, 12% [95% CI 6%–22%]). There were 145 children with CSF-positive infections addressed by 2017 IDSA guideline recommendations, with 135 at risk for reinfection. No reinfections were observed among children whose treatment was fully adherent (0/3, 0% [95% CI 0%–64%]), and 18 reinfections were observed among those whose infection treatment was nonadherent (18/132, 14% [95% CI 8%–21%]).

CONCLUSIONS

There is no clear evidence that either adherence to IDSA guidelines or duration of treatment longer than recommended is associated with reduction in reinfection rates. Because IDSA guidelines recommend shorter IV antibiotic durations than are typically used, improvement efforts to reduce IV antibiotic use in CSF shunt infection treatment can and should utilize IDSA guidelines.

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C. Michael Honey, Zurab Ivanishvili, Christopher R. Honey and Manraj K. S. Heran

OBJECTIVE

The location of the human spinothalamic tract (STT) in the anterolateral spinal cord has been known for more than a century. The exact nature of the neuronal fiber lamination within the STT, however, remains controversial. After correlating in vivo macrostimulation-induced pain/temperature sensation during percutaneous cervical cordotomy with simultaneous CT imaging of the electrode tip location, the authors present a modern description of the somatotopy of the human cervical STT.

METHODS

Twenty patients underwent CT-guided percutaneous cervical cordotomy to alleviate contralateral medication-refractory cancer pain. Patient responses to electrical stimulation (0.01–0.1 V, 50 Hz, 1 msec) were recorded and the electrode location for each response was documented with a contemporaneous CT scan. In a post hoc analysis of the data, the location for each patient’s response(s) was measured and drawn on a diagram of their cord. Positive responses were represented only when the lowest possible voltage (≤ 0.02 V) elicited a response. Negative responses were recorded if there was no clinical response at 0.1 V.

RESULTS

Clinically, patients did well with an average reduction in opiates of 75% at 1 week, and 67% were able to leave the palliative care unit. The size of the cervical cord varied between patients, with an average lateral extent (width) of 11 mm and a height of 9 mm. Responses from the lower limb were represented superficially (lateral) and posteriorly within the anterolateral cord. The area with responses from the upper limb was larger and surrounded those with responses from the lower limb primarily anteriorly and medially, but also posteriorly.

CONCLUSIONS

In this study, the somatotopic organization of the human STT was elucidated for the first time using in vivo macrostimulation and contemporaneous CT imaging during cordotomy. In this cohort of patients, the STT from the lower-limb region was located superficially and posteriorly in the anterolateral quadrant of the cervical cord, with the STT from the upper-limb region surrounding it primarily anteriorly and medially (deep) but also posteriorly. The authors discuss how the previous methods of cordotomy may have biased the earlier versions of STT lamination. They suggest that an ideal spinal cord entry site for cordotomy of either the upper- or lower-limb pain fibers is halfway between the equator and anterior pole of the cord.

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Karthik Madhavan, Lee Onn Chieng, Valerie L. Armstrong and Michael Y. Wang

OBJECTIVE

Discitis and osteomyelitis are seen in end-stage renal disease (ESRD) patients due to repeated vascular access for hemodialysis and urinary tract infections leading to recurrent bacteremia. Discitis and osteomyelitis are underdiagnosed due to the nonspecific initial presentation of back pain. In this article, we review the literature for better understanding of the problem and the importance of early diagnosis by primary care physicians and nephrologists. In addition, we discuss the decision-making, follow-up, management, and neurological outcomes.

METHODS

A detailed PubMed search was performed using the following terms: “end stage renal disease (ESRD)” and “chronic renal failure (CRF),” combined with “spine infections,” “spondylodiscitis,” “discitis,” and “osteomyelitis.” Search results were limited to articles written in English, case reports, and case series from 1973 to 2012. Editorials, reviews, and commentaries were excluded. Only studies involving human patients were included. The authors also included 4 patients from their own patient population.

RESULTS

A total of 30 articles met the inclusion criteria. Including the 4 patients from the authors’ patient population, 212 patients with spine infections and maintenance dialysis were identified. The patients’ ages ranged from 38 to 78 years. The duration of dialysis ranged from a few days to 16 years. The time from onset of back pain to diagnosis ranged from 3 days to 6 months. The most common causative organism was Staphylococcus aureus, followed by Staphylococcus epidermidis and gram-negative bacteria. Most of the patients were treated with antibiotics alone (76.8%), although surgery was indicated when patients presented with neurological deficits (p < 0.011). Approximately one-quarter of the patients developed neurological deficits, with devastating consequences. Fever and neurological deficits at presentation, culture positive for methicillin-resistant S. aureus, and age > 65 years were highly correlated with mortality in our analysis.

CONCLUSIONS

Several risk factors lead to failure of antibiotics and progression of disease in patients with ESRD. Challenges to diagnosis include vague presenting symptoms, co-existing destructive spondyloarthropathy, poor immune response, chronic elevations of inflammatory markers, and recurrent bacteremia. Infectious processes are more likely to cause permanent neurological deficits than transient deficits. The authors recommend close observation and serial imaging of these patients for early signs of neurological deficits. Any signs of disease progression will require aggressive surgical debridement.

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Dan Laukka, Melissa Rahi, Riitta Parkkola, Tero Vahlberg, Arttu Rintala, Eeva Salo and Jaakko Rinne

OBJECTIVE

Kawasaki disease (KD) is a vasculitis that can cause aneurysm formation in coronary arteries and, more rarely, in peripheral arteries. A possible connection between KD and intracranial aneurysms is unclear. The purpose of this study was to determine if KD is associated with intracranial aneurysms.

METHODS

In this prospective cohort study, all patients hospitalized and diagnosed with KD in the authors’ hospital district area in the period from 1978 to 1995 were identified. Patients with a current age ≥ 25 years and a history of KD in childhood were included in the study, which was conducted between 2016 and 2017. Magnetic resonance angiography (MRA) of the brain was performed in all patients.

RESULTS

Forty patients (25 males), whose mean age was 33.5 ± 3.9 years (mean ± standard deviation), were eligible for study inclusion. The mean age at KD diagnosis was 3.9 ± 3.1 years, and the mean follow-up was 29.5 ± 4.3 years. Six patients (15%) had coronary arterial lesions during the acute illness of KD. None of the patients (0%) had intracranial aneurysms on brain MRA, which is significantly under the prevalence of 10% (95% CI 0%–8.8%, p = 0.03) that is the recommended limit for intracranial aneurysm screening.

CONCLUSIONS

The study results suggest that KD is not associated with an increased prevalence of intracranial aneurysms and that screening for intracranial aneurysms is not warranted in patients with a history of KD.