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Junming Ma, Jianru Xiao and Liangzhe Wang

The authors describe the case of a 44-year-old man with multilevel lumbar spinal Rosai–Dorfman disease (RDD), a rare clinical entity. To the authors' knowledge, there have been only 2 cases of lumbar spinal involvement of RDD (epidural) reported in the literature, and the current case is the third but the only one showing lumbar spinal intradural involvement of RDD. This case of RDD mimicked a meningioma both clinically and radiologically. The patient underwent a procedure in which the tumor was excised, and postoperatively the patient made a clinically acceptable recovery. Vertebral canal involvement of RDD should be considered in the differential diagnosis of vertebral canal tumors. Resection is an acceptable treatment option.

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Honglin Teng, John Hsiang, Chunlei Wu, Meihao Wang, Haifeng Wei, Xinghai Yang and Jianru Xiao

Object

The authors propose an easy MR imaging method to measure and categorize individual anatomical variations within the cervicothoracic junction (CTJ). Furthermore, they propose guidelines for selection of the appropriate approach based on this new categorization system.

Methods

In the midsagittal section of the cervicothoracic MR imaging studies obtained in 95 Chinese patients, a triangle was drawn among 3 points: the suprasternal notch (SSN), the midpoint of the anterior border of the C7/T1 intervertebral disc, and the corresponding anterior border in the CTJ at the level of the SSN. The angle above the SSN was specified as the cervicothoracic angle (CTA). The spatial position between the brachiocephalic vein (BCV), the aortic arch, and the CTA was also measured. Based on these measurements involving the CTA, 3 different patient-specific categorizations are proposed to assist surgeons with selection of the appropriate anterior approach to the CTJ. Three categories of operative approach based on whether the most caudal part of the lesion site was above, within, or below the area of the CTA were classified. The patients were divided into long- or short-necked groups based on whether their own CTA was greater than (long necked) or less than (short necked) the average CTA. Finally, a left BCV was called superiorly located when it coursed above the manubrium. The method was evaluated in 21 patients with spinal bone tumors in the CTJ to illustrate the measurement of both the CTA and the great vessels, and corresponding approach selections.

Results

In this series of 95 patients, the most common vertebra above the SSN was T-3, especially the upper one-third of T-3. The mean CTA was 47.64°. The left BCV was superior to the manubrium in 21.1% of the 95 cases, and 93.6% of the left BCVs were at the T-2 and T-3 levels. Type A and most Type B lesions could be addressed via a low suprasternal approach, or this approach combined with manubriotomy, if necessary. Type C lesions falling below the CTA will need alternative exposure techniques, including manubriotomy, sternotomy, lateral extracavitary, or thoracotomy. The spinal levels that could be exposed in the long-necked CTJ group were always 1 or 2 vertebral levels lower than those in the short-necked CTJ group during the anterior low suprasternal approach without the manubriotomy.

Conclusions

Imaging of the thoracic manubrium should be routinely included on MR imaging studies obtained in the CTJ. It is important for the surgeon to understand the pertinent anatomy of the individual patients and to determine the feasible surgical approaches after evaluating the CTA and vascular factors preoperatively. An anterior low suprasternal approach, or this approach combined with manubriotomy, is applicable in most of the cases in the CTJ. It should be cautioned that preoperatively unrecognized variations of the left BCV above the SSN might result in potential intraoperative trauma during an anterior approach.

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Junming Ma, Zhipeng Wu, Xinghai Yang and Jianru Xiao

Object

The goal in this study was to retrospectively investigate the clinical efficacy of surgical treatment for cervical dystrophic kyphotic deformity due to neurofibromatosis Type 1.

Methods

Between January 1998 and July 2008, 8 patients with cervical dystrophic kyphotic deformity due to neurofibromatosis Type 1 (mean Cobb angle of 58.5°) were surgically treated in the authors' department. The mean age at surgery was 19 years (range 12–38 years). Among these patients, 1 with a Cobb angle of 52° and good flexibility underwent single anterior correction, whereas the other 7 patients with severe deformity and poor flexibility received combined anterior and posterior cervical osteotomy. Motor-evoked potential studies were used intraoperatively for spinal cord monitoring. Radiographic assessment and Japanese Orthopaedic Association scoring were used to evaluate the clinical outcome.

Results

No severe neurological complications were noted. Two patients complained of persistent neck and shoulder pain after combined anterior and posterior correction, which alleviated after conservative treatment half a year later. All patients were followed up for a mean of 21.1 months (range 6–36 months). All patients had a solid bone fusion at the latest follow-up, with Japanese Orthopaedic Association scoring improving from 11.5 preoperatively to 14.1 postoperatively (p < 0.01) at the final follow-up. The kyphotic deformities improved significantly, with average Cobb angles of 2.5° postoperatively and 4.1° at final follow-up.

Conclusions

The deformity of neurofibromatosis with cervical kyphosis is severe, and surgery carries a high risk of failure. Although premature fusion may be performed, the deformity may still progress, and this situation may lead to failure of surgery. The successful management of this disease requires early recognition and a more aggressive and reliable intervention to prevent disastrous worsening of the deformity. Meticulous preoperative evaluation, appropriate surgical strategy, and skilled technique were essential for successful surgical treatment and good clinical results.

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Yu Wang, Jianru Xiao, Zhipeng Wu, Quan Huang, Wending Huang, Qing Zhu, Zaijun Lin and Liangzhe Wang

Object

Cervical chordomas are rare lesions and usually bring about challenges in treatment planning because of their wide extension and complicated adjacent anatomy. There are few large published series at present focusing on cervical chordomas. The authors studied a consecutive series of 14 patients with primary cervical chordomas who underwent surgical treatment and were observed between 1989 and 2008. By reviewing the clinical patterns and follow-up data, they sought to investigate the clinical characters, tailor the appropriate surgical techniques, and establish prognosis factors for cervical chordomas.

Methods

Hospitalization and follow-up data in the 14 patients were collected. All patients underwent piecemeal tumor excision and reconstruction for stability; total spondylectomy was achieved in 5 cases. Postoperative radiotherapy was administered in all patients. Kaplan-Meier plots were used to represent tumor recurrence and patient survival, and log-rank testing was used to determine the risk factors of local recurrence.

Results

Follow-up ranged from 8 to 120 months (mean 58.6 months). Symptom and neural status in most patients improved after surgery. The 1- and 5-year disease-free survival rates were 78.6% and 50%, respectively, and the 1- and 5-year survival rates were 92.9% and 85.7%, respectively. Log-rank tests revealed that the following variables were significantly associated with a high rate of tumor recurrence: age less than 40 years or greater than 70 years (p = 0.006) and an upper cervical tumor location (p = 0.019).

Conclusions

Chordomas in the cervical spine are usually neoplasms that exhibit insidious growth and a wide extension by the time of diagnosis. Radical intralesional debulking surgery and postoperative radiotherapy have been effective treatment. A limited application of en bloc tumor resection and the highly likely intraoperative intralesional tumor seeding may partially explain the high local recurrence rate, whereas the chance of distant metastases, fortunately, is very low. Most recurrence were documented within 3 years. Some specific surgical techniques should be emphasized to minimize tumor seeding. Patients with upper cervical chordomas, younger adults, and elderly adults have worse prognosis. For patients with chordoma extending to both the anterior and posterior spinal columns, total spondylectomy combined with piecemeal excision is recommended for a better prognosis.

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Qing Zhu, Ming Qian, Jianru Xiao, Zhipeng Wu, Yu Wang and Jishen Zhang

Object

Calcified meningiomas are an uncommon type of meningioma. This study details the clinical features, treatment, and follow-up of 11 calcified meningiomas treated from 2002 to 2009, for the purpose of providing general information, describing the skill required for the surgery, and detailing the imaging study of these tumors.

Methods

Between 2002 and 2009, 11 patients underwent surgery for the treatment of calcified meningiomas. All were treated by the same group of doctors at the same institution, including surgery and rehabilitation after surgery. The minimum 3-year (> 36 months) follow-up data from the 11 patients were detailed. Neurological function was evaluated twice, based on the Frankel scale and Japanese Orthopaedic Association scoring system. The first evaluation occurred before surgery and the second 3 years after surgery.

Results

In 3 cases, the Frankel score decreased by 1 level. In a comparison of the duration of preoperative symptoms, age, degree of canal stenosis, and intraoperative blood loss, it was found that the greater the degree of canal stenosis, the poorer the outcome of the patient. Calcified meningiomas were more likely to adhere to the nerves and dura, a finding that might explain the high incidence of neurological dysfunction and CSF leakage after surgery.

Conclusions

Calcified meningiomas are the most rare of all meningiomas. It appears that a greater degree of canal stenosis can lead to a poorer outcome. Computed tomography scans and MRI with contrast enhancement are recommended for intraspinal tumors before surgery to exclude the possibility of calcification. For calcified meningiomas, precise tumor resection, dura repair during surgery, and medical care after surgery are important for achieving an acceptable outcome.

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Hsuan-Kan Chang, Jau-Ching Wu, Diego Shih-Chieh Lin, Chih-Chang Chang, Tsung-Hsi Tu, Wen-Cheng Huang and Henrich Cheng

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Wei Xu, Yu Wang, Jing Wang, Xinghai Yang, Weibo Liu, Wang Zhou, Tielong Liu and Jianru Xiao

OBJECTIVE

The objective of this study was to investigate the effect of long-term bisphosphonate treatment on reducing local recurrence of sacral giant cell tumors (GCTs) after nerve-sparing surgery.

METHODS

Thirty-five consecutive patients with sacral GCTs who received treatment in Shanghai Changzheng Hospital between January 2000 and December 2010 were included in this study. Between January 2007 and December 2010, 19 patients received bisphosphonates following nerve-sparing surgery. Before January 2007, 16 patients received nerve-sparing surgery alone, and these cases were included as the control group. The difference in clinical data between the groups was compared by Student's t-test and 2-tailed chi-square or Fisher's exact test. The postoperative recurrence-free survival (RFS) and overall survival (OS) rates were estimated by the Kaplan-Meier method and compared between the groups by log-rank test. A p value < 0.05 was considered statistically significant.

RESULTS

All of the patients had relatively good nerve function. The clinical data were homogeneous between the groups. The local recurrence rate was 10.53% (2 of 19) in the bisphosphonate treatment group and 43.75% (7 of 16) in the control group. The log-rank test showed that the 3-year RFS and 3-year OS in the bisphosphonate treatment group were significantly higher than those in the control group (RFS 89.5% vs 56.3%, p = 0.04; OS 100% vs 81.3%, p = 0.05).

CONCLUSIONS

The long-term use of bisphosphonates after nerve-sparing surgery is a viable option for the treatment of sacral GCTs. This approach could reduce local recurrences while preserving nerve function.

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Qi Jia, Xin Gao, Zhenhua Zhou, Bin Lan, Jian Zhao, Tielong Liu, Xinghai Yang, Haifeng Wei and Jianru Xiao

OBJECTIVE

Spinal instability or neurological impairment caused by spinal brown tumors (BTs) with end-stage renal disease (ESRD) is an acute condition that needs urgent surgery. There is not much published information on BTs of the mobile spine given the rarity of the disease, and the literature shows inconsistent treatment options and ambiguous follow-up information. The aim of the present study was to elucidate the clinical features, anesthesia management, and surgical treatment for this rare disease through long-term follow-up observations.

METHODS

Clinical, laboratory, radiological, and perioperative data on 6 consecutive patients with spinal BTs who had been admitted to the authors’ institution between 2010 and 2016 were retrospectively reviewed. The literature on spinal BT with secondary hyperparathyroidism was also reviewed. Summaries of the clinical features and anesthesia management are provided.

RESULTS

The mean age of the 6 patients was 45.5 years (range 35–62 years). Lesions were located in the cervical segment (2 cases) and thoracic segment (4 cases). Localized pain was the most common complaint, and pathological fracture occurred in 5 patients. Elevation of serum alkaline phosphate (AKP) and parathyroid hormone (PTH) was a common phenomenon. Four patients underwent circumferential resection and 2 underwent laminectomy, with parathyroidectomy performed in all patients. The anesthesia process was uneventful for all patients. The mean follow-up was 33 months (range 26–40 months). No spinal lesion progression occurred in any patient. The Karnofsky Performance Status score improved to 80–90 by 3 months after surgery.

CONCLUSIONS

Although uncommon, spinal BTs should be a diagnostic consideration in patients with ESRD. The thoracic spine is the most frequently affected site. ESRD is not a contraindication for surgery; with the assistance of experienced anesthesiologists, urgent surgery is the preferred option to alleviate neurological impairment and restore spinal stability.

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Shaohui He, Yuduo Xu, Jialin Li, Yue Zhang, Haifeng Wei and Jianru Xiao

OBJECTIVE

Leiomyogenic tumor of the spine is rare with limited published information. Here, the authors report the clinical features and long-term surgical outcomes and investigate the prognostic factors affecting disease-free survival (DFS).

METHODS

Twelve patients presented to the authors’ institution for surgical treatment from January 2005 to December 2018. The clinical characteristics and outcomes were retrospectively reviewed, and the DFS rate was estimated using the Kaplan-Meier method. The log-rank test was used to identify the potential prognostic factors, with p < 0.05 considered statistically significant.

RESULTS

The mean patient age was 49.7 ± 12.9 years (range 22–73 years). Four patients underwent marginal en bloc resection, and 8 patients underwent conventional piecemeal resection. Pathological diagnosis revealed leiomyosarcoma in 9 patients and leiomyoma in 3 patients. Three patients had tumor recurrence at a mean follow-up of 10.4 months (range 7.0–15.0 months), while 4 developed metastases at an average of 13.8 months (range 5.5–21.3 months) postoperatively. During the mean follow-up of 33.7 months (range 9.6–78.5 months), the estimated 1- and 5-year DFS rates were 66.7% and 38.2%, respectively. Albumin loss > 20 g/L after surgery, Ki-67 positivity > 10%, and piecemeal resection were correlated with worse DFS.

CONCLUSIONS

Surgical management of spinal leiomyogenic tumors is challenging due to the high rate of recurrence and metastases. En bloc resection should be performed in eligible patients. Albumin loss > 20 g/L and the Ki-67 index may be independent factors affecting prognosis.

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Shaohui He, Chen Ye, Nanzhe Zhong, Minglei Yang, Xinghai Yang and Jianru Xiao

OBJECTIVE

The surgical treatment of an upper cervical spinal tumor (UCST) at C1–2/C1–3 is challenging due to anterior exposure and reconstruction. Limited information has been published concerning the effective approach and reconstruction for an anterior procedure after C1–2/C1–3 UCST resection. The authors attempted to introduce a novel, customized, anterior craniocervical reconstruction between the occipital condyles and inferior vertebrae through a modified high-cervical retropharyngeal approach (mHCRA) in addressing C1–2/C1–3 spinal tumors.

METHODS

Seven consecutive patients underwent 2-stage UCST resection with circumferential reconstruction. Posterior decompression and occiput-cervical instrumentation was conducted at the stage 1 operation, and anterior craniocervical reconstruction using a 3D-printed implant was performed between the occipital condyles and inferior vertebrae via an mHCRA. The clinical characteristics, perioperative complications, and radiological outcomes were reviewed, and the rationale for anterior craniocervical reconstruction was also clarified.

RESULTS

The mean age of the 7 patients in the study was 47.6 ± 19.0 years (range 12–72 years) when referred to the authors’ center. Six patients (85.7%) had recurrent tumor status, and the interval from primary to recurrence status was 53.0 ± 33.7 months (range 24–105 months). Four patients (57.1%) were diagnosed with a spinal tumor involving C1–3, and 3 patients (42.9%) with a C1–2 tumor. For the anterior procedure, the mean surgical duration and average blood loss were 4.1 ± 0.9 hours (range 3.0–6.0 hours) and 558.3 ± 400.5 ml (range 100–1300 ml), respectively. No severe perioperative complications occurred, except 1 patient with transient dysphagia. The mean pre- and postoperative visual analog scale scores were 8.0 ± 0.8 (range 7–9) and 2.4 ± 0.5 (range 2.0–3.0; p < 0.001), respectively, and the mean improvement rate of cervical spinal cord function was 54.7% ± 13.8% (range 42.9%–83.3%) based on the modified Japanese Orthopaedic Association scale score (p < 0.001). Circumferential instrumentation was in good position and no evidence of disease was found at the mean follow-up of 14.8 months (range 7.3–24.2 months).

CONCLUSIONS

The mHCRA provides optimal access to the surgical field at the C0–3 level. Customized anterior craniocervical fixation between the occipital condyles and inferior vertebrae can be feasible and effective in managing anterior reconstruction after UCST resection.