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Ziya L. Gokaslan, Patricia L. Zadnik, Daniel M. Sciubba, Niccole Germscheid, C. Rory Goodwin, Jean-Paul Wolinsky, Chetan Bettegowda, Mari L. Groves, Alessandro Luzzati, Laurence D. Rhines, Charles G. Fisher, Peter Pal Varga, Mark B. Dekutoski, Michelle J. Clarke, Michael G. Fehlings, Nasir A. Quraishi, Dean Chou, Jeremy J. Reynolds, Richard P. Williams, Norio Kawahara and Stefano Boriani

OBJECT

A chordoma is an indolent primary spinal tumor that has devastating effects on the patient's life. These lesions are chemoresistant, resistant to conventional radiotherapy, and moderately sensitive to proton therapy; however, en bloc resection remains the preferred treatment for optimizing patient outcomes. While multiple small and largely retrospective studies have investigated the outcomes following en bloc resection of chordomas in the sacrum, there have been few large-scale studies on patients with chordomas of the mobile spine. The goal of this study was to review the outcomes of surgically treated patients with mobile spine chordomas at multiple international centers with respect to local recurrence and survival. This multiinstitutional retrospective study collected data between 1988 and 2012 about prognosis-predicting factors, including various clinical characteristics and surgical techniques for mobile spine chordoma. Tumors were classified according to the Enneking principles and analyzed in 2 treatment cohorts: Enneking-appropriate (EA) and Enneking-inappropriate (EI) cohorts. Patients were categorized as EA when the final pathological assessment of the margin matched the Enneking recommendation; otherwise, they were categorized as EI.

METHODS

Descriptive statistics were used to summarize the data (Student t-test, chi-square, and Fisher exact tests). Recurrence and survival data were analyzed using Kaplan-Meier survival curves, log-rank tests, and multivariate Cox proportional hazard modeling.

RESULTS

A total of 166 patients (55 female and 111 male patients) with mobile spine chordoma were included. The median patient follow-up was 2.6 years (range 1 day to 22.5 years). Fifty-eight (41%) patients were EA and 84 (59%) patients were EI. The type of biopsy (p < 0.001), spinal location (p = 0.018), and if the patient received adjuvant therapy (p < 0.001) were significantly different between the 2 cohorts. Overall, 58 (35%) patients developed local recurrence and 57 (34%) patients died. Median survival was 7.0 years postoperative: 8.4 years postoperative for EA patients and 6.4 years postoperative for EI patients (p = 0.023). The multivariate analysis showed that the EI cohort was significantly associated with an increased risk of local recurrence in comparison with the EA cohort (HR 7.02; 95% CI 2.96–16.6; p < 0.001), although no significant difference in survival was observed.

CONCLUSIONS

EA resection plays a major role in decreasing the risk for local recurrence in patients with chordoma of the mobile spine.

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Jennifer E. Kim, John Pang, Joani M. Christensen, Devin Coon, Patricia L. Zadnik, Jean-Paul Wolinsky, Ziya L. Gokaslan, Ali Bydon, Daniel M. Sciubba, Timothy Witham, Richard J. Redett and Justin M. Sacks

OBJECT

Total en bloc sacrectomy is a dramatic procedure that results in extensive sacral defects. The authors present a series of patients who underwent flap reconstruction after total sacrectomy, report clinical outcomes, and provide a treatment algorithm to guide surgical care of this unique patient population.

METHODS

After institutional review board approval, data were collected for all patients who underwent total sacrectomy between 2002 and 2012 at The Johns Hopkins Hospital. Variables included demographic data, medical history, tumor characteristics, surgical details, postoperative complications, and clinical outcomes. All subtotal sacrectomies were excluded.

RESULTS

Between 2002 and 2012, 9 patients underwent total sacrectomy with flap reconstruction. Diagnoses included chordoma (n = 5), osteoblastoma (n = 1), sarcoma (n = 2), and metastatic colon cancer (n = 1). Six patients received gluteus maximus (GM) flaps with a prosthetic rectal sling following a single-stage, posterior sacrectomy. Four required additional paraspinous muscle (PSM) or pedicled latissimus dorsi (LD) fasciocutaneous flaps. Three patients underwent multistage sacrectomy with an anterior-posterior approach, 2 of whom received pedicled vertical rectus abdominis myocutaneous (VRAM) flaps, and 1 of whom received local GM, LD, and PSM flaps. Flap complications included dehiscence (n = 4) and infection (n = 1). During the 1st year of follow-up, 2 of 9 patients (22%) were able to ambulate with an assistive device by the 1st postoperative month, and 6 of 9 (67%) were ambulatory with a walker by the 3rd postoperative month. By postoperative Month 12, 5 of 9 patients (56%)—or 5 of 5 patients not lost to follow-up (100%)—were able to able to ambulate independently.

CONCLUSIONS

The authors' experience suggests that the GM and pedicled VRAM flaps are reliable options for softtissue reconstruction of total sacrectomy defects. For posterior-only operations, GM flaps with or without a prosthetic rectal sling are generally used. For multistage operations including a laparotomy, the authors consider the pedicled VRAM flap to be the gold standard for simultaneous reconstruction of the pelvic diaphragm and obliteration of dead space.

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Patricia L. Zadnik, C. Rory Goodwin, Kristophe J. Karami, Ankit I. Mehta, Anubhav G. Amin, Mari L. Groves, Jean-Paul Wolinsky, Timothy F. Witham, Ali Bydon, Ziya L. Gokaslan and Daniel M. Sciubba

OBJECT

Multiple myeloma is the most common primary tumor of the spine and is the most common primary malignant tumor of bone. Although spinal myeloma is classically a radiosensitive lesion, clinical or radiographic signs of instability merit surgical intervention. The authors present the epidemiology, surgical indications, and outcome data of a series of consecutive cases involving 31 surgically treated patients with diagnoses of multiple myeloma and plasmacytoma of the spine (the largest such series reported to date).

METHODS

Surgical instability was the criterion for operative intervention in this patient cohort. The Spinal Instability Neoplastic Score (SINS) was used to make this assessment of instability. The cases were analyzed using location of the lesion, spinal levels involved, Frankel score, adjuvant therapy, functional outcome, and patient survival.

RESULTS

All patients undergoing surgical intervention were determined to have indeterminate or gross spinal column instability according to SINS criteria. The median survival was 78.9 months. No significant difference in survival was seen for patients with higher SINS scores or for older patients (> 55 years). There was a statistically significant difference in survival benefit observed for patients receiving chemotherapy and radiation versus radiation alone as an adjuvant to surgery (p = 0.02).

CONCLUSIONS

In this 10-year analysis, the authors report outcomes of surgical intervention for patients with indeterminate or gross spinal instability due to multiple myeloma and plasmacytoma of the spine with improved neurological function following surgery and low rates of instrumentation failure.

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Michelle J. Clarke, Patricia L. Zadnik, Mari L. Groves, Daniel M. Sciubba, Timothy F. Witham, Ali Bydon, Ziya L. Gokaslan and Jean-Paul Wolinsky

OBJECT

Recently, aggressive surgical techniques and a push toward en bloc resections of certain tumors have resulted in a need for creative spinal column reconstruction. Iatrogenic instability following these resections requires a thoughtful approach to adequately transfer load-bearing forces from the skull and upper cervical spine to the subaxial spine.

METHODS

The authors present a series of 7 cases in which lateral mass reconstruction with a cage or fibular strut graft was used to provide load-bearing support, including 1 case of bilateral cage placement.

RESULTS

The authors discuss the surgical nuances of en bloc resection of high cervical tumors and explain their technique for lateral mass cage placement. Additionally, they provide their rationale for the use of these constructs throughout the craniocervical junction and subaxial spine.

CONCLUSIONS

Lateral mass reconstruction provides a potential alternative or adjuvant method of restoring the load-bearing capabilities of the cervical spine.

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Michelle J. Clarke, Patricia L. Zadnik, Mari L. Groves, Hormuzdiyar H. Dasenbrock, Daniel M. Sciubba, Wesley Hsu, Timothy F. Witham, Ali Bydon, Ziya L. Gokaslan and Jean-Paul Wolinsky

Object

Traditionally, hemisacrectomy and internal hemipelvectomy procedures have required both an anterior and a posterior approach. A posterior-only approach has the potential to complete an en bloc tumor resection and spinopelvic reconstruction while reducing surgical morbidity.

Methods

The authors describe 3 cases in which en bloc resection of the hemisacrum and ilium and subsequent lumbopelvic and pelvic ring reconstruction were performed from a posterior-only approach. Two more traditional anterior and posterior staged procedures are also included for comparison.

Results

In all 3 cases, an oncologically appropriate surgery and spinopelvic reconstruction were performed through a posterior-only approach.

Conclusions

The advantage of a midline posterior approach is the ability to perform a lumbosacral reconstruction, necessary in cases in which the S-1 body is iatrogenically disrupted during tumor resection.

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Camilo A. Molina, Christopher P. Ames, Dean Chou, Laurence D. Rhines, Patrick C. Hsieh, Patricia L. Zadnik, Jean-Paul Wolinsky, Ziya L. Gokaslan and Daniel M. Sciubba

Object

Chordomas involving the mobile spine are ideally managed via en bloc resection with reconstruction to optimize local control and possibly offer cure. In the cervical spine, local anatomy poses unique challenges, limiting the feasibility of aggressive resection. The authors present a multi-institutional series of 16 cases of cervical chordomas removed en bloc. Particular attention was paid to clinical outcome, complications, and recurrence. In addition, outcomes were assessed according to position of tumor at the C1–2 level versus the subaxial (SA) spine (C3–7).

Methods

The authors reviewed cases involving patients who underwent en bloc resection of cervical chordoma at 4 large spine centers. Patients were included if the lesion epicenter involved the C-1 to C-7 vertebral bodies. Demographic data and details of surgery, follow-up course, exposure to adjuvant therapy, and complications were obtained. Outcome was correlated with presence of tumor in C1–2 versus subaxial spine via a Student t-test.

Results

Sixteen patients were identified (mean age at presentation 55 ± 14 years). Seven cases (44%) cases involved C1–2, and 16 involved the subaxial spine. Median survival did not differ significantly different between the C1–2 (72 months) and SA (60 months) groups (p = 0.65). A combined (staged anteroposterior) approach was used in 81% of the cases. Use of the combined approach was significantly more common in treatment of subaxial than C1–2 tumors (100% vs 57%, p = 0.04). En bloc resection was attempted via an anterior approach in 6% of cases (C1–2: 14.3%; SA: 0%; p = 0.17) and a posterior approach in 13% of cases (C1–2: 29%; SA: 0%; p = 0.09). The most commonly reported margin classification was marginal (56% of cases), followed by violated (25%) and wide (19%). En bloc excision of subaxial tumors was significantly more likely to result in marginal margins than excision of C1–2 tumors (C1–2: 29%; SA: 78%; p = 0.03). C1–2 tumors were associated with significantly higher rates of postoperative complications (C1–2: 71%; SA: 22%; p = 0.03). Both local and distant tumor recurrence was greatest for C1–2 tumors (local C1–2: 29%; local SA: 11%; distant C1–2: 14%; distant SA: 0%). Statistical analysis of tumor recurrence based on tumor location was not possible due to the small number of cases. There was no between-groups difference in exposure to postoperative adjuvant radiotherapy. There was no difference in median survival between groups receiving proton beam radiotherapy or intensity-modulated radiotherapy versus no radiation therapy (p = 0.8).

Conclusions

Compared with en bloc resection of chordomas involving the subaxial cervical spine, en bloc resection of chordomas involving the upper cervical spine (C1–2) is associated with poorer outcomes, such as less favorable margins, higher rates of complications, and increased tumor recurrence. Data from this cohort do not support a statistically significant difference in survival for patients with C1–2 versus subaxial disease, but larger studies are needed to further study survival differences.

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Paul E. Kaloostian, Patricia L. Zadnik, Jennifer E. Kim, Mari L. Groves, Jean-Paul Wolinsky, Ziya L. Gokaslan, Timothy F. Witham, Ali Bydon and Daniel M. Sciubba

Pheochromocytomas of the spine are uncommon and require careful preoperative planning. The authors retrospectively reviewed the charts of 5 patients with metastatic spinal pheochromocytoma who had undergone surgical treatment over the past 10 years at their medical center. They reviewed patient age, history of pheochromocytoma resection, extent and location of metastases, history of alpha blockage, surgical level, surgical procedure, postoperative complications, tumor recurrence, and survival. Metastases involved the cervical (1 patient), thoracic (3 patients), and lumbar (2 patients) levels. Preoperative treatment included primary pheochromocytoma resection, chemotherapy, alpha blockade, embolization, and radiation. Three patients had tumor recurrence, and 2 underwent 2-stage reoperations for tumor extension. Hemodynamic complications were common: 2 patients developed pulseless electrical activity arrest within 4 months after surgery, 1 patient had profound postoperative tachycardia with fever and an elevated creatine kinase level, and 1 patient experienced transient postoperative hypotension and paraplegia. One patient died of complications related to disseminated cerebral and spinal disease.

With careful preoperative and surgical management, patients with symptomatic metastatic spinal pheochromocytoma can benefit from aggressive surgical treatment. Postoperative cardiovascular complications are common even months after surgery, and patients should be closely monitored long term.

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Paul E. Kaloostian, Patricia L. Zadnik, Ahmed J. Awad, Edward McCarthy, Jean-Paul Wolinsky and Daniel M. Sciubba

Resection of metastatic pheochromocytomas may be complicated by transient postoperative neurological deficits due to hypotension. The authors report the first case of en bloc excision of a spinal pheochromocytoma with associated long-term hypertensive management off all medication. Interestingly, this is the first case of transient hypotension following en bloc resection of pheochromocytoma associated with temporary hypotension-associated neurological decline that resolved completely after correction of hypotension postoperatively. A 23-year-old man with a prior adrenalectomy for pheochromocytoma presented with focal thoracic pain. He had a known T-10 vertebral body lesion for which he received chemotherapy and radiation therapy. Imaging demonstrated increased destruction of the T-10 vertebral body, which was concerning for tumor growth. The patient underwent angiographic embolization followed by single-stage posterior en bloc vertebrectomy with placement of a cage and posterior instrumentation and fusion without event. However, approximately 24 hours after surgery, the patient's systolic blood pressure was consistently no higher than 70 mm Hg. During this time, he began suffering from severe bilateral lower-extremity weakness. His systolic blood pressure increased with dopamine, and his strength immediately improved. The patient's oral regimen of adrenergic blockade was stopped, and he recovered without event. Since that time, the patient has been symptom free and requires no antihypertensive medication. The role of en bloc resection for metastatic lesions of the spine is controversial but may be warranted in cases of metastatic pheochromocytoma. En bloc resection avoids intralesional tumor resection and thus may help prevent complications of hypertensive crisis associated with hormonal secretion and extensive blood loss, which are not uncommon with pheochromocytoma resection surgeries. Additionally, the role of en bloc spondylectomy in this setting may allow for metabolic treatment as patients with actively secreting tumors may no longer require antiadrenergic medications.