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Ian E. McCutcheon, Keith E. Friend, Tammy M. Gerdes, Bing-Mei Zhang, David M. Wildrick and Gregory N. Fuller

Object. Although human meningioma cells have been heterotopically implanted in nude mice, introducing these cells into intracranial locations seems more likely to reproduce normal patterns of tumor growth. To provide an orthotopic xenograft model of meningioma, the authors implanted a controlled quantity of meningioma cells at subdural and intracerebral sites in athymic mice.

Methods. Malignant (one tumor), atypical (two tumors), or benign (three tumors) meningiomas were placed into primary cell cultures. Cells (106/10 µl) from these cultures and from an immortalized malignant meningioma cell line, IOMM-Lee, were injected with stereotactic guidance into the frontal white matter or subdural space of athymic mice. Survival curves were plotted for mice receiving tumor cells of each histological type and according to injection site. Other mice were killed at intervals and their heads were sectioned whole. Hematoxylin and eosin staining of these sections revealed the extent of tumor growth.

Conclusions. The median length of survival for mice with malignant, atypical, or benign tumors was 19, 42, or longer than 84 days, respectively. Atypical and malignant tumors were invasive, but did not metastasize extracranially. Malignant tumors uniformly showed leptomeningeal dissemination and those implanted intracerebrally grew locally and spread noncontiguously to the ventricles, choroid plexus, convexities, and skull base. Tumors formed in only 50% of mice injected with benign meningioma cells, whereas injection of more aggressive cells was uniformly successful at tumor production. The three types of human meningiomas grown intracranially in athymic mice maintained their relative positions in the spectrum of malignancy. However, atypical meningiomas became more aggressive after xenografting and acquired malignant features, implying that there had been immune constraint in the original host. Tumor cells injected into brain parenchyma migrated to more optimal environments and grew best there. This model provides insights into the biology of meningiomas and may be useful for testing new therapies.

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Scott L. Zuckerman, Ganesh Rao, Laurence D. Rhines, Ian E. McCutcheon, Richard G. Everson and Claudio E. Tatsui

OBJECTIVE

Treatment of epidural spinal cord compression (ESCC) caused by tumor includes surgical decompression and stabilization followed by postoperative radiation. In the case of severe axial loading impairment, anterior column reconstruction is indicated. The authors describe the use of interbody distraction to restore vertebral body height and correct kyphotic angulation prior to reconstruction with polymethylmethacrylate (PMMA), and report the long-term durability of such reconstruction.

METHODS

A single institution, prospective series of patients with ESCC undergoing single-stage decompression, anterior column reconstruction, and posterior instrumentation from 2013 to 2016 was retrospectively analyzed. Several demographic, perioperative, and radiographic measurements were collected. Descriptive statistics were compiled, in addition to postoperative changes in anterior height, posterior height, and kyphosis. Paired Student t-tests were performed for each variable. Overall survival was calculated using the techniques described by Kaplan and Meier.

RESULTS

Twenty-one patients underwent single-stage posterior decompression with interbody distraction and anterior column reconstruction using PMMA. The median age and Karnofsky Performance Scale score were 61 years and 70, respectively. Primary tumors included renal cell (n = 8), lung (n = 4), multiple myeloma (n = 2), prostate (n = 2), and other (n = 5). Eighteen patients underwent a single-level vertebral body reconstruction and 3 underwent multilevel transpedicular corpectomies. The median survival duration was 13.3 months. In the immediate postoperative setting, statistically significant improvement was noted in anterior body height (p = 0.0017, 95% confidence interval [CI] −4.15 to −1.11) and posterior body height (p = 0.0116, 95% CI −3.14 to −0.45) in all patients, and improved kyphosis was observed in those with oblique endplates (p = 0.0002, 95% CI 11.16–20.27). In the median follow-up duration of 13.9 months, the authors observed 3 cases of asymptomatic PMMA subsidence. One patient required reoperation in the form of extension of fusion.

CONCLUSIONS

In situ interbody distraction allows safe and durable reconstruction with PMMA, restores vertebral height, and corrects kyphotic deformities associated with severe pathological fractures caused by tumor. This is accomplished with minimal manipulation of the thecal sac and avoiding an extensive 360° surgical approach in patients who cannot tolerate extensive surgery.

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Sherise D. Ferguson, Nicholas B. Levine, Dima Suki, Andrew J. Tsung, Fredrick F. Lang, Raymond Sawaya, Jeffrey S. Weinberg and Ian E. McCutcheon

OBJECTIVE

Fourth ventricle tumors are rare, and surgical series are typically small, comprising a single pathology, or focused exclusively on pediatric populations. This study investigated surgical outcome and complications following fourth ventricle tumor resection in a diverse patient population. This is the largest cohort of fourth ventricle tumors described in the literature to date.

METHODS

This is an 18-year (1993–2010) retrospective review of 55 cases involving patients undergoing surgery for tumors of the fourth ventricle. Data included patient demographic characteristics, pathological and radiographic tumor characteristics, and surgical factors (approach, surgical adjuncts, extent of resection, etc.). The neurological and medical complications following resection were collected and outcomes at 30 days, 90 days, 6 months, and 1 year were reviewed to determine patient recovery. Patient, tumor, and surgical factors were analyzed to determine factors associated with the frequently encountered postoperative neurological complications.

RESULTS

There were no postoperative deaths. Gross-total resection was achieved in 75% of cases. Forty-five percent of patients experienced at least 1 major neurological complication, while 31% had minor complications only. New or worsening gait/focal motor disturbance (56%), speech/swallowing deficits (38%), and cranial nerve deficits (31%) were the most common neurological deficits in the immediate postoperative period. Of these, cranial nerve deficits were the least likely to resolve at follow-up. Multivariate analysis showed that patients undergoing a transvermian approach had a higher incidence of postoperative cranial nerve deficits, gait disturbance, and speech/swallowing deficits than those treated with a telovelar approach. The use of surgical adjuncts (intraoperative navigation, neurophysiological monitoring) did not significantly affect neurological outcome. Twenty-two percent of patients required postoperative CSF diversion following tumor resection. Patients who required intraoperative ventriculostomy, those undergoing a transvermian approach, and pediatric patients (< 18 years old) were all more likely to require postoperative CSF diversion. Twenty percent of patients suffered at least 1 medical complication following tumor resection. Most complications were respiratory, with the most common being postoperative respiratory failure (14%), followed by pneumonia (13%).

CONCLUSIONS

The occurrence of complications after fourth ventricle tumor surgery is not rare. Postoperative neurological sequelae were frequent, but a substantial number of patients had neurological improvement at long-term followup. Of the neurological complications analyzed, postoperative cranial nerve deficits were the least likely to completely resolve at follow-up. Of all the patient, tumor, and surgical variables included in the analysis, surgical approach had the most significant impact on neurological morbidity, with the telovelar approach being associated with less morbidity.

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Stephen J. Hentschel, Laurence D. Rhines, Franklin C. Wong, Ziya L. Gokaslan and Ian E. McCutcheon

Object. Little has been written about the appropriate diagnosis, investigation, and management of subarachnoid—pleural fistula (SPF). The authors report a series of patients with SPF that developed after resection of spinal tumor and discuss the diagnosis and treatment of this entity.

Methods. Between 1993 and 2002, nine patients with SPF observed after spinal surgery at the M. D. Anderson Cancer Center were prospectively followed. In all patients the tumors were located in the thoracic region, and the most common entity was vertebral body metastasis (six cases), with renal cell carcinoma being the most common form of the disease (three cases). All but one patient underwent surgery via a transthoracic approach; in only one patient an intradural approach was performed. The most common presentation was overt cerebrospinal fluid (CSF) leakage, manifesting as chest tube drainage (four cases) or as leakage through the wound (one case). A definitive diagnosis of SPF was established in four patients, with evidence of extraspinal leakage on an 111In-radionuclide CSF study. Although all patients initially underwent a trial of lumbar CSF drainage, all but one required open repair, including creation of intercostal muscle (three cases) and omental (one case) flaps.

Conclusions. After spinal surgery in which the thorax is entered, a diagnosis of SPF should be considered in any patient with abnormal chest tube output, persistent pleural effusion, or clinical evidence of intracranial hypotension. The diagnosis should be confirmed by performing a radionuclide-labeled CSF study. Definitive open repair is required in most cases and preferentially consists of a vascularized tissue graft, which is most easily obtained from an intercostal muscle flap.

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John R. Floyd II, Elizabeth R. Keeler, Elizabeth D. Euscher and Ian E. McCutcheon

Sciatic (catamenial) radiculopathy, waxing and waning with the menstrual cycle, is an uncommon condition typically caused by pelvic endometriosis affecting the lumbosacral plexus or proximal sciatic nerve. The authors describe a woman with catamenial sciatica caused by endometriosis affecting the sciatic nerve trunk in the upper thigh. Symptomatic with leg pain for 5 years, this patient developed gluteal atrophy and sensory loss and decreased strength in the L-5 dermatomyotome, a distribution confirmed by electromyography. Magnetic resonance imaging suggested thickening of the sciatic nerve at and distal to the sciatic notch. At operation the nerve showed extrinsic and intrinsic abnormality, proven to be endometriosis. Her symptoms improved, and she began gonadotropin-releasing hormone agonist therapy for further suppression. This very unusual case shows that endometriosis can affect the sciatic nerve over a range of territory inside and outside the pelvis, and that surgery must be appropriately directed to avoid negative exploration. Surgical decompression achieves good relief of symptoms, and medical therapy also allows sustained suppression of this disease.

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Christopher M. McPherson, Dima Suki, Ian E. McCutcheon, Ziya L. Gokaslan, Laurence D. Rhines and Ehud Mendel

Object.

Metastastic lesions have been reported in 5 to 40% of patients with spinal and sacrococcygeal chordoma, but few contemporary series of chordoma metastastic disease exist in the literature. Additionally, the outcome in patients with chordoma-induced metastastic neoplasms remains unclear. The authors performed a retrospective review of the neurosurgery database at the University of Texas M. D. Anderson Cancer Center in Houston to determine the incidence of metastatic disease in a contemporary series of spinal and sacrococcygeal chordoma as well as to determine the outcomes.

Methods.

Thirty-seven patients underwent surgery for spinal and sacrococcygeal chordoma between June 1, 1993, and March 31, 2004. All records were reviewed, and appropriate statistical analyses were used to compare patient data for preoperative characteristics, treatments, and outcomes.

The authors identified seven patients (19%) in whom metastatic disease developed; in three the disease had metastasized to the lungs only, in two to the lungs and liver, and in two to distant locations in the spine. There were no significant differences in age, sex, tumor location, or history of radiation treatments between patients with and those without metastases. In cases with local recurrent tumors, metastastic disease was more likely to develop than in those without recurrence (28 compared with 0%, respectively; p = 0.07). In two (12%) of 17 patients who underwent en bloc resection, metastatic disease developed, whereas it developed in five (25%) of 20 patients treated by curettage (p = 0.42). The median time from first surgery to the appearance of metastatic disease, as calculated using the Kaplan–Meier method, was 143.4 months (95% confidence interval [CI] 66.8–219.9). The median survival duration of patients with metastatic disease after the first surgery was 106 months (95% CI 55.7–155.7), and this did not differ significantly from that in patients in whom no metastases developed (p = 0.93).

Conclusions.

Spinal chordoma metastasized to other locations in 19% of the patients in this series. In patients with local disease recurrence, metastatic lesions are more likely to develop. Metastatic lesions were shown to be aggressive in some cases. Surgery and chemotherapy can play a role in controlling metastatic disease.

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Daryl R. Fourney, Dima Abi-Said, Laurence D. Rhines, Garrett L. Walsh, Frederick F. Lang, Ian E. McCutcheon and Ziya L. Gokaslan

Object. Thoracic or lumbar spine malignant tumors involving both the anterior and posterior columns represent a complex surgical problem. The authors review the results of treating patients with these lesions in whom surgery was performed via a simultaneous anterior—posterior approach.

Methods. The hospital records of 26 patients who underwent surgery via simultaneous combined approach for thoracic and lumbar spinal tumors at our institution from July 1994 to March 2000 were reviewed. Surgery was performed with the patients in the lateral decubitus position for the procedure. The technical details are reported.

The mean survival determined by Kaplan—Meier analysis was 43.4 months for the 15 patients with primary malignant tumors and 22.5 months for the 11 patients with metastatic spinal disease. At 1 month after surgery, 23 (96%) of 24 patients who complained of pain preoperatively reported improvements (p < 0.001, Wilcoxon signed-rank test), and eight (62%) of 13 patients with preoperative neurological deficits were functionally improved (p = 0.01). There were nine major complications, five minor complications, and no deaths within 30 days of surgery. Two patients (8%) later underwent surgery for recurrent tumor.

Conclusions. The simultaneous anterior—posterior approach is a safe and feasible alternative for the exposure tumors of the thoracic and lumbar spine that involve both the anterior and posterior columns. Advantages of the approach include direct visualization of adjacent neurovascular structures, the ability to achieve complete resection of lesions involving all three columns simultaneously (optimizing hemostasis), and the ability to perform excellent dorsal and ventral stabilization in one operative session.

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Edward W. Akeyson, Ian E. McCutcheon, Mark A. Pershouse, Peter A. Steck and Gregory N. Fuller

✓ The authors describe a malignant peripheral primitive neuroectodermal tumor (PNET) that originated in the median nerve in an elderly adult. After the diagnosis was made by biopsy, the patient underwent radical local resection with interpositional vein grafting of the brachial artery. The tumor had the typical appearance of a primitive neural tumor with small, round cells forming rosettes. It stained positively for both the Ewing's sarcoma/peripheral PNET antigen (HBA-71) and neuron-specific enolase, confirming its neural origin. Ultrastructural examination revealed dense core granules and suggested neural differentiation of the neoplasm. Cytogenetic analysis suggested a chromosome (11;22) translocation typical of peripheral PNET. Early reports consisted of tumors arising solely in peripheral nerves, but recent series have focused mainly on tumors arising in the soft tissues other than nerves. There are no other cases of true PNET of peripheral nerve in the modern literature that have been fully characterized by immunohistochemical, ultrastructural, and cytogenetic criteria. Although peripheral PNETs occur more commonly in children, this unusual neoplasm should be considered in the differential diagnosis of peripheral nerve neoplasms in adults. Early diagnosis is desirable because of its aggressive nature and poor outcome.

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Hassan H. Amhaz, Roukoz B. Chamoun, Steven G. Waguespack, Komal Shah and Ian E. McCutcheon

Rathke cleft cysts (RCCs) are benign cystic lesions of the sella that arise from the remnants of Rathke pouch. Although most are asymptomatic, symptoms can result from mass effect and commonly include headache, endocrinopathy, or visual field disturbance. Although asymptomatic patients undergo conservative treatment, patients with symptoms are typically treated surgically. The authors report 9 patients with symptomatic cystic sellar lesions and imaging characteristics consistent with an RCC; in all cases there was spontaneous involution of the lesions, and in 5 of 7 patients presenting with headache the symptom resolved. Spontaneous involution of an RCC may be more common than the paucity of prior reports would suggest, especially because the natural history of both symptomatic and asymptomatic RCCs is poorly understood. The potential for spontaneous involution, together with the clinical course of the patients reported here, supports a conservative approach for patients with symptomatic RCCs presenting solely with headache.