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  • Author or Editor: Laurence D. Rhines x
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Anne L. Versteeg, Nicolas Dea, Stefano Boriani, Peter P. Varga, Alessandro Luzzati, Michael G. Fehlings, Mark H. Bilsky, Laurence D. Rhines, Jeremy J. Reynolds, Mark. B. Dekutoski, Ziya L. Gokaslan, Niccole M. Germscheid and Charles G. Fisher

OBJECTIVE

Osteoblastoma is a rare primary benign bone tumor with a predilection for the spinal column. Although of benign origin, osteoblastomas tend to behave more aggressively clinically than other benign tumors. Because of the low incidence of osteoblastomas, evidence-based treatment guidelines and high-quality research are lacking, which has resulted in inconsistent treatment. The goal of this study was to determine whether application of the Enneking classification in the management of spinal osteoblastomas influences local recurrence and survival time.

METHODS

A multicenter database of patients who underwent surgical intervention for spinal osteoblastoma was developed by the AOSpine Knowledge Forum Tumor. Patient data pertaining to demographics, diagnosis, treatment, cross-sectional survival, and local recurrence were collected. Patients in 2 cohorts, based on the Enneking classification of the tumor (Enneking appropriate [EA] and Enneking inappropriate [EI]), were analyzed. If the final pathology margin matched the Enneking-recommended surgical margin, the tumor was classified as EA; if not, it was classified as EI.

RESULTS

A total of 102 patients diagnosed with a spinal osteoblastoma were identified between November 1991 and June 2012. Twenty-nine patients were omitted from the analysis because of short follow-up time, incomplete survival data, or invalid staging, which left 73 patients for the final analysis. Thirteen (18%) patients suffered a local recurrence, and 6 (8%) patients died during the study period. Local recurrence was strongly associated with mortality (relative risk 9.2; p = 0.008). When adjusted for Enneking appropriateness, this result was not altered significantly. No significant differences were found between the EA and EI groups in regard to local recurrence and mortality.

CONCLUSIONS

In this evaluation of the largest multicenter cohort of spinal osteoblastomas, local recurrence was found to be strongly associated with mortality. Application of the Enneking classification as a treatment guide for preventing local recurrence was not validated.

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Daniel M. Sciubba, Mohamed Macki, Mohamad Bydon, Niccole M. Germscheid, Jean-Paul Wolinsky, Stefano Boriani, Chetan Bettegowda, Dean Chou, Alessandro Luzzati, Jeremy J. Reynolds, Zsolt Szövérfi, Patti Zadnik, Laurence D. Rhines, Ziya L. Gokaslan, Charles G. Fisher and Peter Paul Varga

OBJECT

Clinical outcomes in patients with primary spinal osteochondromas are limited to small series and sporadic case reports. The authors present data on the first long-term investigation of spinal osteochondroma cases.

METHODS

An international, multicenter ambispective study on primary spinal osteochondroma was performed. Patients were included if they were diagnosed with an osteochondroma of the spine and received surgical treatment between October 1996 and June 2012 with at least 1 follow-up. Perioperative prognostic variables, including patient age, tumor size, spinal level, and resection, were analyzed in reference to long-term local recurrence and survival. Tumor resections were compared using Enneking appropriate (EA) or Enneking inappropriate surgical margins.

RESULTS

Osteochondromas were diagnosed in 27 patients at an average age of 37 years. Twenty-two lesions were found in the mobile spine (cervical, thoracic, or lumbar) and 5 in the fixed spine (sacrum). Twenty-three cases (88%) were benign tumors (Enneking tumor Stages 1–3), whereas 3 (12%) exhibited malignant changes (Enneking tumor Stages IA–IIB). Sixteen patients (62%) underwent en bloc treatment—that is, wide or marginal resection—and 10 (38%) underwent intralesional resection. Twenty-four operations (92%) followed EA margins. No one received adjuvant therapy. Two patients (8%) experienced recurrences: one in the fixed spine and one in the mobile spine. Both recurrences occurred in latent Stage 1 tumors following en bloc resection. No osteochondroma-related deaths were observed.

CONCLUSIONS

In the present study, most patients underwent en bloc resection and were treated as EA cases. Both recurrences occurred in the Stage 1 tumor cohort. Therefore, although benign in character, osteochondromas still require careful management and thorough follow-up.

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Dean Chou, Mark H. Bilsky, Alessandro Luzzati, Charles G. Fisher, Ziya L. Gokaslan, Laurence D. Rhines, Mark B. Dekutoski, Michael G. Fehlings, Ravi Ghag, Peter Varga, Stefano Boriani, Niccole M. Germscheid, Jeremy J. Reynolds and the AOSpine Knowledge Forum Tumor

OBJECTIVE

Malignant peripheral nerve sheath tumors (MPNSTs) are rare soft-tissue sarcomas. Resection is the mainstay of treatment and the most important prognostic factor. However, complete resection of spinal MPNSTs with tumor-free margins is challenging due to the likelihood of residual tumor cells. The objective of this study was to describe whether the type of Enneking resection in the management of spinal MPNSTs had an effect on local recurrence and survival.

METHODS

The AOSpine Knowledge Forum Tumor developed a multicenter database that includes demographic, diagnostic, therapeutic, local recurrence, and survival data on patients with primary spinal column tumors. Patients who had undergone surgery for a primary spinal MPNST were included and were analyzed in 2 groups: 1) those undergoing Enneking appropriate (EA) resections and 2) those undergoing Enneking inappropriate (EI) resections. EA surgery was performed if there was histopathological evidence of an intact tumor pseudocapsule and at least a marginal resection on a vital structure. EI surgery was performed if there was an intentional or inadvertent transgression of the margin.

RESULTS

Between 1993 and 2012, 29 primary spine MPNSTs were identified in 12 (41%) females and 17 (59%) males with a mean age at diagnosis of 40 ± 17 years (range 5–74 years). The median patient follow-up was 1.3 years (range 42 days to 11.2 years). In total, 14 (48%) patients died and 14 (48%) patients suffered a local recurrence, 10 (71%) of whom died. Within 2 years after surgery, the median survival and local recurrence were not achieved. Data about Enneking appropriateness of surgery were available for 27 patients; 9 (33%) underwent an EA procedure and 18 (67%) underwent an EI procedure. Enneking appropriateness did not have a significant influence on local recurrence or survival. Twenty-two patients underwent adjuvant treatment with combined chemo- and radiotherapy (n = 7), chemotherapy alone (n = 3), or radiotherapy alone (n = 12). Adjuvant therapy had no significant influence on recurrence or survival.

CONCLUSIONS

The rates of recurrence and survival were similar for spinal MPNSTs regardless of whether patients had an EA or EI resection or received adjuvant therapy. Other factors such as variability of pathologist interpretation, PET CT correlation, or neurofibromatosis Type 1 status may play a role in patient outcome. Nonetheless, MPNSTs should still be treated as sarcomas until further evidence is known. The authors recommend an individualized approach with careful multidisciplinary decision making, and the patient should be informed about the morbidity of en bloc surgery when considering MPNST resection.

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Mark B. Dekutoski, Michelle J. Clarke, Peter Rose, Alessandro Luzzati, Laurence D. Rhines, Peter P. Varga, Charles G. Fisher, Dean Chou, Michael G. Fehlings, Jeremy J. Reynolds, Richard Williams, Nasir A. Quraishi, Niccole M. Germscheid, Daniel M. Sciubba, Ziya L. Gokaslan, Stefano Boriani and The AOSpine Knowledge Forum Tumor

OBJECTIVE

Primary spinal osteosarcomas are rare and aggressive neoplasms. Poor outcomes can occur, as obtaining marginal margins is technically demanding; further Enneking-appropriate en bloc resection can have significant morbidity. The goal of this study is to identify prognostic variables for local recurrence and mortality in surgically treated patients diagnosed with a primary osteosarcoma of the spine.

METHODS

A multicenter ambispective database of surgically treated patients with primary spine osteosarcomas was developed by AOSpine Knowledge Forum Tumor. Patient demographic, diagnosis, treatment, perioperative morbidity, local recurrence, and cross-sectional survival data were collected. Tumors were classified in 2 cohorts: Enneking appropriate (EA) and Enneking inappropriate (EI), as defined by pathology margin matching Enneking-recommended surgical margins. Prognostic variables were analyzed in reference to local recurrence and survival.

RESULTS

Between 1987 and 2012, 58 patients (32 female patients) underwent surgical treatment for primary spinal osteosarcoma. Patients were followed for a mean period of 3.5 ± 3.5 years (range 0.5 days to 14.3 years). The median survival for the entire cohort was 6.7 years postoperative. Twenty-four (41%) patients died, and 17 (30%) patients suffered a local recurrence, 10 (59%) of whom died. Twenty-nine (53%) patients underwent EA resection while 26 (47%) patients underwent EI resection with a postoperative median survival of 6.8 and 3.7 years, respectively (p = 0.048). EI patients had a higher rate of local recurrence than EA patients (p = 0.001). Patient age, previous surgery, biopsy type, tumor size, spine level, and chemotherapy timing did not significantly influence recurrence and survival.

CONCLUSIONS

Osteosarcoma of the spine presents a significant challenge, and most patients die in spite of aggressive surgery. There is a significant decrease in recurrence and an increase in survival with en bloc resection (EA) when compared with intralesional resection (EI). The effect of adjuvant and neoadjuvant chemotherapeutics, as well as method of biopsy, requires further exploration.

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Nicolas Dea, Charles G. Fisher, Jeremy J. Reynolds, Joseph H. Schwab, Laurence D. Rhines, Ziya L. Gokaslan, Chetan Bettegowda, Arjun Sahgal, Áron Lazáry, Alessandro Luzzati, Stefano Boriani, Alessandro Gasbarrini, Ilya Laufer, Raphaële Charest-Morin, Feng Wei, William Teixeira, Niccole M. Germscheid, Francis J. Hornicek, Thomas F. DeLaney, John H. Shin and the AOSpine Knowledge Forum Tumor

OBJECTIVE

The purpose of this study was to investigate the spectrum of current treatment protocols for managing newly diagnosed chordoma of the mobile spine and sacrum.

METHODS

A survey on the treatment of spinal chordoma was distributed electronically to members of the AOSpine Knowledge Forum Tumor, including neurosurgeons, orthopedic surgeons, and radiation oncologists from North America, South America, Europe, Asia, and Australia. Survey participants were pre-identified clinicians from centers with expertise in the treatment of spinal tumors. The suvey responses were analyzed using descriptive statistics.

RESULTS

Thirty-nine of 43 (91%) participants completed the survey. Most (80%) indicated that they favor en bloc resection without preoperative neoadjuvant radiation therapy (RT) when en bloc resection is feasible with acceptable morbidity. The main area of disagreement was with the role of postoperative RT, where 41% preferred giving RT only if positive margins were achieved and 38% preferred giving RT irrespective of margin status. When en bloc resection would result in significant morbidity, 33% preferred planned intralesional resection followed by RT, and 33% preferred giving neoadjuvant RT prior to surgery. In total, 8 treatment protocols were identified: 3 in which en bloc resection is feasible with acceptable morbidity and 5 in which en bloc resection would result in significant morbidity.

CONCLUSIONS

The results confirm that there is treatment variability across centers worldwide for managing newly diagnosed chordoma of the mobile spine and sacrum. This information will be used to design an international prospective cohort study to determine the most appropriate treatment strategy for patients with spinal chordoma.