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Michael M. Safaee, Cecilia L. Dalle Ore, Corinna C. Zygourakis, Vedat Deviren and Christopher P. Ames

OBJECTIVE

Proximal junctional kyphosis (PJK) is a well-recognized complication of surgery for adult spinal deformity and is characterized by increased kyphosis at the upper instrumented vertebra (UIV). PJK prevention strategies have the potential to decrease morbidity and cost by reducing rates of proximal junctional failure (PJF), which the authors define as radiographic PJK plus clinical sequelae requiring revision surgery.

METHODS

The authors performed an analysis of 195 consecutive patients with adult spinal deformity. Age, sex, levels fused, upper instrumented vertebra (UIV), use of 3-column osteotomy, pelvic fixation, and mean time to follow-up were collected. The authors also reviewed operative reports to assess for the use of surgical adjuncts targeted toward PJK prevention, including ligament augmentation, hook fixation, and vertebroplasty. The cost of surgery, including direct and total costs, was also assessed at index surgery and revision surgery. Only revision surgery for PJF was included.

RESULTS

The mean age of the cohort was 64 years (range 25–84 years); 135 (69%) patients were female. The mean number of levels fused was 10 (range 2–18) with the UIV as follows: 2 cervical (1%), 73 upper thoracic (37%), 108 lower thoracic (55%), and 12 lumbar (6%). Ligament augmentation was used in 99 cases (51%), hook fixation in 60 cases (31%), and vertebroplasty in 71 cases (36%). PJF occurred in 18 cases (9%). Univariate analysis found that ligament augmentation and hook fixation were associated with decreased rates of PJF. However, in a multivariate model that also incorporated age, sex, and UIV, only ligament augmentation maintained a significant association with PJF reduction (OR 0.196, 95% CI 0.050–0.774; p = 0.020). Patients with ligament augmentation, compared with those without, had a higher cost of index surgery, but ligament augmentation was overall cost effective and produced significant cost savings. In sensitivity analyses in which we independently varied the reduction in PJF, cost of ligament augmentation, and cost of reoperation by ± 50%, ligament augmentation remained a cost-effective strategy for PJF prevention.

CONCLUSIONS

Prevention strategies for PJK/PJF are limited, and their cost-effectiveness has yet to be established. The authors present the results of 195 patients with adult spinal deformity and show that ligament augmentation is associated with significant reductions in PJF in both univariate and multivariate analyses, and that this intervention is cost-effective. Future studies will need to determine if these clinical results are reproducible, but for high-risk cases, these data suggest an important role of ligament augmentation for PJF prevention and cost savings.

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William B. Feldman, Aaron J. Clark, Michael Safaee, Christopher P. Ames and Andrew T. Parsa

Object

Myxopapillary ependymomas (MPEs) are rare WHO Grade I tumors found in the conus medullaris, cauda equina, and filum terminale. Treatment generally consists of resection with or without adjuvant radiotherapy. Evidence-based guidelines for surgical management are lacking due to the rarity of this tumor.

Methods

An English-language PubMed search was performed using the key words “myxopapillary” and “ependymoma.” Reports describing fewer than 3 patients or those lacking data on the extent of resection or radiotherapy were excluded. A total of 28 articles describing 475 patients met the authors' inclusion criteria. Patients were grouped by extent of resection and whether or not they underwent adjuvant radiotherapy. Differences in recurrence rates were assessed by chi-square test.

Results

The overall recurrence rate was 15.5% in patients treated by gross-total resection (GTR) and 32.6% in patients treated by subtotal resection (STR), irrespective of whether they underwent adjuvant therapy (p < 0.001). Regardless of the extent of resection, adjuvant radiotherapy was not associated with a decrease in recurrence rates. The overall recurrence rate was 15.6% in patients who underwent GTR and radiotherapy compared with 15.9% in patients who underwent GTR alone (p = 0.58), and it was 29.3% in patients who underwent STR and radiotherapy compared with 35.1% in those who underwent STR alone (p = 0.53). The difference between recurrence rates for patients who underwent GTR alone versus STR and radiotherapy was statistically significant (p = 0.02). Subgroup analysis demonstrated significantly higher recurrence rates in pediatric patients compared with adults (40.5% vs 23.4%, respectively; p = 0.02). Even in the setting of GTR alone, recurrence rates were higher in pediatric patients (65% vs 7.6%; p < 0.001).

Conclusions

Gross-total resection alone is associated with decreased recurrence rates compared with STR with or without radiotherapy. The authors' results suggest that treatment goals should include attempted GTR whenever possible. The observation that children benefitted from radiation therapy to a greater extent than did adults suggests that biological differences between tumors in these patient populations warrants more rigorous scientific studies.

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Cecilia L. Dalle Ore, Darryl Lau, Jessica L. Davis, Michael M. Safaee and Christopher P. Ames

Juvenile ossifying fibroma (JOF) is a rare benign bone tumor that occurs most frequently in the craniofacial bones of children and young adults. There are few case reports that describe its involvement outside the craniofacial skeleton, especially within the spinal column. While JOF is classified as a benign lesion, it may be locally aggressive and demonstrate a high propensity for recurrence, even after resection. Definitive surgical management may be challenging in naive cases, but it is particularly challenging in recurrent cases and when extensive spinal reconstruction is warranted. In this report, the authors describe the diagnosis and surgical management of a 29-year-old man who presented with a large recurrent sacral trabecular-subtype JOF. A review of literature regarding JOFs, management of recurrent primary spinal tumors, and sacral reconstruction are discussed.

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Michael M. Safaee, Cecilia L. Dalle Ore, Corinna C. Zygourakis, Vedat Deviren and Christopher P. Ames

OBJECTIVE

Bone morphogenetic protein (BMP) is associated with reduced rates of pseudarthrosis and has the potential to decrease the need for revision surgery. There are limited data evaluating the cost-benefit of BMP for pseudarthrosis-related prevention surgery in adult spinal deformity.

METHODS

The authors performed a single-center retrospective review of 200 consecutive patients with adult spinal deformity. Demographic data and costs of BMP, primary surgery, and revision surgery for pseudarthrosis were collected. Patients with less than 12 months of follow-up or with infection, tumor, or neuromuscular disease were excluded.

RESULTS

One hundred fifty-one patients (107 [71%] women) with a mean age of 65 years met the inclusion criteria. The mean number of levels fused was 10; BMP was used in 98 cases (65%), and the mean follow-up was 23 months. Fifteen patients (10%) underwent surgical revision for pseudarthrosis; BMP use was associated with an 11% absolute risk reduction in the rate of reoperation (17% vs 6%, p = 0.033), with a number needed to treat of 9.2. There were no significant differences in age, sex, upper instrumented vertebra, or number of levels fused in patients who received BMP. In a multivariate model including age, sex, number of levels fused, and the upper instrumented vertebra, only BMP (OR 0.250, 95% CI 0.078–0.797; p = 0.019) was associated with revision surgery for pseudarthrosis. The mean direct cost of primary surgery was $87,653 ± $19,879, and the mean direct cost of BMP was $10,444 ± $4607. The mean direct cost of revision surgery was $52,153 ± $26,985. The authors independently varied the efficacy of BMP, cost of BMP, and cost of reoperation by ± 50%; only reductions in the cost of BMP resulted in a cost savings per 100 patients. Using these data, the authors estimated a price point of $5663 in order for BMP to be cost-neutral.

CONCLUSIONS

Use of BMP was associated with a significant reduction in the rates of revision surgery for pseudarthrosis. At its current price, the direct in-hospital costs for BMP exceed the costs associated with revision surgery; however, this likely underestimates the true value of BMP when considering the savings associated with reductions in rehabilitation, therapy, medication, and additional outpatient costs.

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Thomas J. Buell, Davis G. Taylor, Ching-Jen Chen, Christopher I. Shaffrey, Justin S. Smith and Shay Bess

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Darryl Lau, Cecilia L. Dalle Ore, Patrick Reid, Michael M. Safaee, Vedat Deviren, Justin S. Smith, Christopher I. Shaffrey and Christopher P. Ames

OBJECTIVE

The benefits and utility of routine neuromonitoring with motor and somatosensory evoked potentials during lumbar spine surgery remain unclear. This study assesses measures of performance and utility of transcranial motor evoked potentials (MEPs) during lumbar pedicle subtraction osteotomy (PSO).

METHODS

This is a retrospective study of a single-surgeon cohort of consecutive adult spinal deformity (ASD) patients who underwent lumbar PSO from 2006 to 2016. A blinded neurophysiologist reviewed individual cases for MEP changes. Multivariate analysis was performed to determine whether changes correlated with neurological deficits. Measures of performance were calculated.

RESULTS

A total of 242 lumbar PSO cases were included. MEP changes occurred in 38 (15.7%) cases; the changes were transient in 21 cases (55.3%) and permanent in 17 (44.7%). Of the patients with permanent changes, 9 (52.9%) had no recovery and 8 (47.1%) had partial recovery of MEP signals. Changes occurred at a mean time of 8.8 minutes following PSO closure (range: during closure to 55 minutes after closure). The mean percentage of MEP signal loss was 72.9%. The overall complication rate was 25.2%, and the incidence of new neurological deficits was 4.1%. On multivariate analysis, MEP signal loss of at least 50% was not associated with complication (p = 0.495) or able to predict postoperative neurological deficits (p = 0.429). Of the 38 cases in which MEP changes were observed, the observation represented a true-positive finding in only 3 cases. Postoperative neurological deficits without MEP changes occurred in 7 cases. Calculated measures of performance were as follows: sensitivity 30.0%, specificity 84.9%, positive predictive value 7.9%, and negative predictive value 96.6%. Regarding the specific characteristics of the MEP changes, only a signal loss of 80% or greater was significantly associated with a higher rate of neurological deficit (23.0% vs 0.0% for loss of less than 80%, p = 0.021); changes of less than 80% were not associated with postoperative deficits.

CONCLUSIONS

Neuromonitoring has a low positive predictive value and low sensitivity for detecting new neurological deficits. Even when neuromonitoring is unchanged, patients can still have new neurological deficits. The utility of transcranial MEP monitoring for lumbar PSO remains unclear but there may be advantages to its use.

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Michael Safaee, Andrew T. Parsa, Nicholas M. Barbaro, Dean Chou, Praveen V. Mummaneni, Philip R. Weinstein, Tarik Tihan and Christopher P. Ames

OBJECT

Intradural extramedullary spine tumors represent two-thirds of all primary spine neoplasms. Approximately half of these are peripheral nerve sheath tumors, mainly neurofibromas and schwannomas. Given the rarity of this disease and, thus, the limited analyses of clinical outcomes, the authors examined the association of tumor location, extent of resection, and neurofibromatosis (NF) status with clinical outcomes.

METHODS

Patients were identified through a search of the University of California, San Francisco, neuropathology database and a separate review of current procedural terminology codes. Data recorded included patient age, patient sex, clinical presentation, presence of NF, tumor type, tumor location, extent of resection (gross-total resection [GTR] or subtotal resection [STR]), and clinical follow-up.

RESULTS

Of 221 tumors in 199 patients (mean age 45 years), 53 were neurofibromas, 163 were schwannomas, and 5 were malignant peripheral nerve sheath tumors. The most common presenting symptom was spinal pain (76%), followed by weakness (36%) and sensory abnormalities (34%). Mean symptom duration was 16 months. In terms of spinal location, neurofibromas were more common in the cervical spine (74% vs 27%, p < 0.001), and schwannomas were more common in the thoracic and lumbosacral spine (73% vs 26%, p < 0.001). Rates of GTR were lower for neurofibromas than schwannomas (51% vs 83%, p < 0.001), regardless of location. Rates of GTR were lower for cervical (54%) than thoracic (90%) and lumbosacral (86%) lesions (p < 0.001). NF was associated with lower rates of GTR among all tumors (43% vs 86%, p < 0.001). The mean follow-up time was 32 months. Recurrence/progression was more common for neurofibromas than schwannomas (17% vs 7%, p = 0.03), although the mean time to recurrence/progression did not differ according to tumor type (45 vs 53 months, p = 0.63). As expected, GTR was associated with lower recurrence rates (4% vs 22%, p < 0.001). According to multivariate analysis, cervical location (OR 0.239, 95% CI 0.110–0.520) and presence of NF (OR 0.166, 95% CI 0.054–0.507) were associated with lower rates of GTR. In a separate model, only GTR (OR 0.141, 95% CI 0.046–0.429) was associated with tumor recurrence.

CONCLUSIONS

Resection is an effective treatment for spinal nerve sheath tumors. Neurofibromas were found more commonly in the cervical spine than in other regions of the spine and were associated with higher rates of recurrence and lower rates of GTR than other tumor types, particularly in patients with NF Types 1 or 2. According to multivariate analysis, both cervical location and presence of NF were associated with lower rates of GTR. According to a second multivariate model, the only variable associated with tumor recurrence was extent of resection. Maximal safe resection remains ideal for these lesions; however, patients with cervical tumors or NF should be counseled about their increased risk for recurrence.

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Michael M. Safaee, Vedat Deviren, Cecilia Dalle Ore, Justin K. Scheer, Darryl Lau, Joseph A. Osorio, Fred Nicholls and Christopher P. Ames

OBJECTIVE

Proximal junctional kyphosis (PJK) is a well-recognized, yet incompletely defined, complication of adult spinal deformity surgery. There is no standardized definition for PJK, but most studies describe PJK as an increase in the proximal junctional angle (PJA) of greater than 10°–20°. Ligament augmentation is a novel strategy for PJK reduction that provides strength to the upper instrumented vertebra (UIV) and adjacent segments while also reducing junctional stress at those levels.

METHODS

In this study, ligament augmentation was used in a consecutive series of adult spinal deformity patients at a single institution. Patient demographics, including age; sex; indication for surgery; revision surgery; surgical approach; and use of 3-column osteotomies, vertebroplasty, or hook fixation at the UIV, were collected. The PJA was measured preoperatively and at last follow-up using 36-inch radiographs. Data on change in PJA and need for revision surgery were collected. Univariate and multivariate analyses were performed to identify factors associated with change in PJA and proximal junctional failure (PJF), defined as PJK requiring surgical correction.

RESULTS

A total of 200 consecutive patients were included: 100 patients before implementation of ligament augmentation and 100 patients after implementation of this technique. The mean age of the ligament augmentation cohort was 66 years, and 67% of patients were women. Over half of these cases (51%) were revision surgeries, with 38% involving a combined anterior or lateral and posterior approach. The mean change in PJA was 6° in the ligament augmentation group compared with 14° in the control group (p < 0.001). Eighty-four patients had a change in PJA of less than 10°. In a multivariate linear regression model, age (p = 0.016), use of hook fixation at the UIV (p = 0.045), and use of ligament augmentation (p < 0.001) were associated with a change in PJA. In a separate model, only ligament augmentation (OR 0.193, p = 0.012) showed a significant association with PJF.

CONCLUSIONS

Ligament augmentation represents a novel technique for the prevention of PJK and PJF. Compared with a well-matched historical cohort, ligament augmentation is associated with a significant decrease in PJK and PJF. These data support the implementation of ligament augmentation in surgery for adult spinal deformity, particularly in patients with a high risk of developing PJK and PJF.

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Michael P. Kelly, Lawrence G. Lenke, Christopher I. Shaffrey, Christopher P. Ames, Leah Y. Carreon, Virginie Lafage, Justin S. Smith and Adam L. Shimer

Object

The goal in this study was to evaluate the risk factors for complications, including new neurological deficits, in the largest cohort of patients with adult spinal deformity to date.

Methods

The Scoli-RISK-1 inclusion criteria were used to identify eligible patients from 5 centers who were treated between June 1, 2009, and June 1, 2011. Records were reviewed for patient demographic information, surgical data, and reports of perioperative complications. Neurological deficits were recorded as preexisting or as new deficits. Patients who underwent 3-column osteotomies (3COs) were compared with those who did not (posterior spinal fusion [PSF]). Between-group comparisons were performed using independent samples t-tests and chi-square analyses.

Results

Two hundred seven patients were identified—75 who underwent PSF and 132 treated with 3CO. In the latter group, patients were older (58.9 vs 49.4 years, p < 0.001), had a higher body mass index (29.0 vs 25.8, p = 0.029), smaller preoperative coronal Cobb measurements (33.8° vs 56.4°, p < 0.001), more preoperative sagittal malalignment (11.7 cm vs 5.4 cm, p < 0.001), and similar sagittal Cobb measurements (45.8° vs 57.7°, p = 0.113). Operating times were similar (393 vs 423 minutes, p = 0.130), although patients in the 3CO group sustained higher estimated blood loss (2120 vs 1700 ml, p = 0.066). Rates of new neurological deficits were similar (PSF: 6.7% vs 3CO: 9.9%, p = 0.389), and rates of any perioperative medical complication were similar (PSF: 46.7% vs 3CO: 50.8%, p = 0.571). Patients who underwent vertebral column resection (VCR) were more likely to sustain medical complications than those treated with pedicle subtraction osteotomy (73.7% vs 46.9%, p = 0.031), although new neurological deficits were similar (15.8% vs 8.8%, p = 0.348). Regression analysis did not reveal significant predictors of neurological injury or complication from collected data.

Conclusions

Despite higher estimated blood loss, rates of all complications (49.3%) and new neurological deficits (8.7%) did not vary for patients who underwent complex reconstruction, whether or not a 3CO was performed. Patients who underwent VCR sustained more medical complications without an increase in new neurological deficits. Prospective studies of patient factors, provider factors, and refined surgical data are needed to define and optimize risk factors for complication and neurological deficits.

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Michael Safaee, Michael C. Oh, Praveen V. Mummaneni, Philip R. Weinstein, Christopher P. Ames, Dean Chou, Mitchel S. Berger, Andrew T. Parsa and Nalin Gupta

Object

Ependymomas are a common type of CNS tumor in children, although only 13% originate from the spinal cord. Aside from location and extent of resection, the factors that affect outcome are not well understood.

Methods

The authors performed a search of an institutional neuropathology database to identify all patients with spinal cord ependymomas treated over the past 20 years. Data on patient age, sex, clinical presentation, symptom duration, tumor location, extent of resection, use of radiation therapy, surgical complications, presence of tumor recurrence, duration of follow-up, and residual symptoms were collected. Pediatric patients were defined as those 21 years of age or younger at diagnosis. The extent of resection was defined by the findings of the postoperative MR images.

Results

A total of 24 pediatric patients with spinal cord ependymomas were identified with the following pathological subtypes: 14 classic (Grade II), 8 myxopapillary (Grade I), and 2 anaplastic (Grade III) ependymomas. Both anaplastic ependymomas originated in the intracranial compartment and spread to the spinal cord at recurrence. The mean follow-up duration for patients with classic and myxopapillary ependymomas was 63 and 45 months, respectively. Seven patients with classic ependymomas underwent gross-total resection (GTR), while 4 received subtotal resection (STR), 2 received STR as well as radiation therapy, and 1 received radiation therapy alone. All but 1 patient with myxopapillary ependymomas underwent GTR. Three recurrences were identified in the Grade II group at 45, 48, and 228 months. A single recurrence was identified in the Grade I group at 71 months. The mean progression-free survival (PFS) was 58 months in the Grade II group and 45 months in the Grade I group.

Conclusions

Extent of resection is an important prognostic factor in all pediatric spinal cord ependymomas, particularly Grade II ependymomas. These data suggest that achieving GTR is more difficult in the upper spinal cord, making tumor location another important factor. Although classified as Grade I lesions, myxopapillary ependymomas had similar outcomes when compared with classic (Grade II) ependymomas, particularly with respect to PFS. Long-term complications or new neurological deficits were rare. Among patients with long-term follow-up, those who underwent GTR had a recurrence rate of 20% compared with 40% among those with STR or biopsy only, suggesting that extent of resection is perhaps a more important prognostic factor than histological grade in predicting PFS, which has been suggested by other data in the literature. Given the relative paucity of these lesions, collaborative multiinstitutional studies are needed, and such efforts should also focus on molecular and genetic analysis to refine the current classification system.