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. 2016 Jul 7;8(2):171–178. doi: 10.1111/os.12234

En Bloc Resection of Primary Malignant Bone Tumor in the Cervical Spine Based on 3‐Dimensional Printing Technology

Jian‐ru Xiao 1,†,✉, Wen‐ding Huang 2,†, Xing‐hai Yang 1,†, Wang‐jun Yan 1, Dian‐wen Song 1, Hai‐feng Wei 1, Tie‐long Liu 1, Zhi‐peng Wu 1, Cheng Yang 1
PMCID: PMC6584397  PMID: 27384725

Abstract

Objective

To investigate the feasibility and safety of en bloc resection of cervical primary malignant bone tumors by a combined anterior and posterior approach based on a three‐dimensional (3‐D) printing model.

Methods

Five patients with primary malignant bone tumors of the cervical spine underwent en bloc resection via a one‐stage combined anteroposterior approach in our hospital from March 2013 to June 2014. They comprised three men and two women of mean age 47.2 years (range, 26–67 years). Three of the tumors were chondrosarcomas and two chordomas. Preoperative 3‐D printing models were created by 3‐D printing technology. Sagittal en bloc resections were planned based on these models and successfully performed. A 360° reconstruction was performed by spinal instrumentation in all cases. Surgical margins, perioperative complications, local control rate and survival rate were assessed.

Results

All patients underwent en bloc excision via a combined posterior and anterior approach in one stage. Mean operative time and estimated blood loss were 465 minutes and 1290 mL, respectively. Mean follow‐up was 21 months. Wide surgical margins were achieved in two patients and marginal resection in three; these three patients underwent postoperative adjuvant radiation therapy. One vertebral artery was ligated and sacrificed in each of three patients. Nerve root involved by tumor was sacrificed in three patients with preoperative upper extremity weakness. One patient (Case 3) had significant transient radiculopathy with paresis postoperatively. Another (Case 4) with C 4 and C 5 chordoma had respiratory difficulties and pneumonia after surgery postoperatively. He recovered completely after 2 weeks’ management with a tracheotomy tube and antibiotics in the intensive care unit. No cerebrovascular complications and wound infection were observed. No local recurrence or instrumentation failure were detected during follow‐up.

Conclusion

Though technically challenging, it is feasible and safe to perform en bloc resection of cervical primary bone tumors. This is the most effective means of managing cervical spine tumors. Preoperative 3‐D printing modelling enables better anatomical understanding of the relationship between the tumor and cervical spine and can assist in planning the surgical procedure.

Keywords: 3D‐printing model, Cervical spine, Complication, En bloc resection, Primary tumor

Introduction

Primary malignant bone tumors of the cervical spine are rare and are associated with significant postoperative morbidity and mortality. Aggressive surgical resection is an important treatment option for neoplasms that are not sensitive to radiotherapy or chemotherapy. Studies have demonstrated that en bloc resection with free margins is the most effective treatment approach, its long‐term disease‐free survival being comparable to that of intralesional techniques1, 2, 3, 4, 5, 6. Boriani et al. retrospectively reviewed a series of studies about primary bone tumors within the mobile spine1, 2, 3. They reported that when margins were tumor free, local control by means of en bloc resection increases to 82% for chondrosarcomas1, 78% for chordomas2 and 92.3% for giant cell tumors3. In the same studies, local control was achieved by intralesional surgery in 0% of chondrosarcomas1, 22% of chordomas2 and 72.2% of giant cell tumors3.

Although total en bloc spondylectomy (TES) for malignant tumors of the thoracic and lumbar spine is well developed and widely performed6, 7, 8, 9, the unique anatomy of the cervical spine makes similar procedures extremely challenging for cervical spinal tumors; in fact, the vertebral arteries (VAs) and cervical nerve roots bilaterally make it impossible to perform TES for these tumors10, 11. Despite these challenges, surgical procedures such as en bloc resection remain the initial form of management for primary cervical spinal tumors1, 2, 3, 4, 5, 12, 13.

With advances in spinal surgery and instrumentation, en bloc resection for cervical spinal tumors has become a viable treatment option. Theoretically, it is the most effective means of achieving local control of primary spinal tumors1, 2, 3, 4, 5, 6, 7, 12, 13. However, various factors hamper performing this procedure in the cervical spine, including the complex anatomy, importance of VAs and cervical nerve roots, and high rate of unfavorable outcomes. Both because of these factors and the limited indications for en bloc resection, only a few reports of en bloc resection of cervical spine tumors have been published10, 11, 12, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27. Fujita et al. first described en bloc resection of a chordoma of the C5 vertebra body with ligation of the left VA and the use of a T‐saw to cut the pedicles via an anterior approach10. However, this was actually an intralesional resection because the involved nerve root was preserved. There have been recent reports en bloc resection of cervical spinal tumors via a combined anterior and posterior approach has been attempted, wide margins being achieved by sacrificing involved VAs and nerve roots14, 15, 18, 19, 20, 23. Additionally, en bloc resection with preservation of the VAs and nerve roots has also been investigated16, 17, 20, 23, 26.

The key point to ensure the success of the procedure is meticulous pre‐operative preparation including planning of the surgical strategy. The advent of three‐dimensional (3‐D) printers has enabled creation of more realistic spinal models that better depict the spinal anatomy28, 29, 30, 31. The most recent techniques for using 3‐D printing models may aid in the planning of complex surgical procedures28, 30, 32, such as en bloc resection of cervical spinal tumors22, thus making their removal easier.

We here describe a technique for complete multilevel cervical en bloc resection with sagittal osteotomy and report the clinical outcomes of five consecutive patients who underwent this procedure. Although en bloc resection is not a novel technique, what is new is that we planned the surgical procedure planning on the basis of a preoperative 3‐D printing plaster model for each patient. The purposes of the article include: (i) discussing the surgical indications and limitations of en bloc resections for cervical spinal tumors; (ii) describing technical details of the operation and procedure‐related complications; and (iii) stressing the importance of preoperative planning with 3‐D printing models.

Materials and Methods

Patients

The inclusion criteria for en bloc resection for cervical spinal tumors were as follows: (i) primary malignant bone tumor; (ii) no involvement of vertebral arteries or functional nerve roots on either side; and (iii) WBB (Weinstein–Boriani–Biagini) staging33 of 4–8/5–9/10–3 sectors and A–D layers. Exclusion criteria included: (i) tumors originating in other regions of the spine; and (ii) any history of other malignancies.

Five eligible patients with cervical spinal tumors who had undergone en bloc resection between March 2013 and June 2014 were retrospectively reviewed from a prospective observational database. Three‐D printing models had been used for surgical planning of the procedures to maximize safe and complete resection of the tumors. This study was approved by the Institutional Review Board of Changzheng Hospital.

Imaging Studies

All patients underwent plain radiography, CT angiograms and MRI to identify the form, location and size of the tumor and its relationship with surrounding structures. All lesions were in the subaxial cervical spine. One vertebral level (C4) was involved in one patient; two vertebral levels in three patients (C6, 7 in two patients and C4, 5 in one); and three levels in one patient (C5–7). Stippled calcification was present on CT images of three patients. The lesions were eccentric in four patients and concentric in one. MRI revealed paravertebral or prevertebral soft tissue masses in all patients. All tumors showed as low‐to‐intermediate signals on T1‐weighted images and as high intensity signals on T2‐weighted sequences with heterogeneous enhancement. Preoperative CT angiograms were performed to determine the patency of involved vessels, caliber and patency of contralateral vessels, and intracranial collateral blood supply to the posterior circulation. The VA and nerve root were encapsulated unilaterally in three cases.

Biopsy

Preoperative biopsy is mandatory for diagnosis of solitary spinal lesions. After imaging studies had been evaluated to ascertain the exact location of the tumor, a biopsy was planned to obtain tissue for a pathological diagnosis. Percutaneous CT‐guided needle or core biopsies are the preferred diagnostic technique in these cases, not only yielding adequate tissue for diagnosis, but also minimizing the possibility of tumor cell contamination of neighboring structures. Four patients underwent needle biopsy under CT guidance and histologic examination showed chondrosarcoma and chordoma, each in two cases. The remaining patient had a local recurrence of chondrosarcoma 8 months after primary surgery.

Surgical Staging

Preoperative CT and MRI contributed to delineating the location and extent of the tumor. In combination with proving the diagnosis by biopsy, surgical staging, including Enneking34 and WBB staging33, was performed on the basis of imaging findings to evaluate the extent of invasion.

3‐D‐Printing Model

Computed tomography angiograms and 3‐D reconstruction were performed after admission to identify details of the anatomical location and structure of the lesion, axial images being reconstructed at 1.0 mm slices with 1.0 mm slice spacing (Brilliance 256 slice CT scanner; Siemens, Sacramento, CA, USA). CT images were processed in DICOM format. After a 3‐D model had been generated, a polylactic acid model in which the “tumor” was colored to differentiate it from the surrounding tissues was printed using a ZPrinter 450; (Z Corporation, Burlington, MA, USA) 24 hours prior to surgery. Because these models were created based on actual patient data, they faithfully reproduced the morphological anatomy of the cervical spine and tumors, enabling surgeons to better understand the dimensions and position of the tumor in relation to adjacent tissues. En bloc resection was planned were made based on the models. They were also used as reference tools during surgery.

Surgical Technique

Step 1: Posterior Approach

In patients in whom unilateral vertebral artery sacrifice was required, preoperative balloon occlusion tests were performed to appraise the feasibility of of such a sacrifice. The 3‐D printing model for the patient was taken into the operating room. The patient was placed prone on the operating table and an X‐ray film taken to confirm the head and neck were in a neutral position. A straight vertical midline incision extending three vertebrae above and below the involved segment(s) was then made over the spinous processes. The paraspinal muscles were dissected subperiosteally from the spinous process, following which the tumor and capsule were meticulously dissected away from the muscle avoiding disruption of the capsule. Pedicle or lateral mass screws (Medtronic Sofamor [Memphis, TN, USA] or DePuy Spine[Raynham, MA, USA]) were placed at least one level above and below the involved vertebra(e). An side rod was put in place to ensure cervical stability after removal of the tumor. Laminectomies were performed at the involved levels according to the 3‐D model. The dura was then carefully dissected from the tumor capsule or pseudocapsule, roots encased by tumor being sacrificed outside its margins. The posterior tubercles of the transverse processes above and below the involved segments were excised to facilitate ligation of the VA.

Step 2: Anterior Approach

After the posterior approach procedure, the patient was positioned supine on the operating table and an especially designed incision made on the affected side of the neck according to the preoperative plan. This incision extended along the anterior border of the sternocleidomastoid muscle down to the necessary level to allow adequate craniocaudal exposure. The entire neurovascular bundle and pharynx were identified and retracted anteriorly with a retractor. The tumor capsule was then identified and the plane between the normal soft tissues and capsule dissected adequately with a safe margin. The 3D models were used to ascertain the relationship between the VA and tumors before exposing them. The involved side VA was freed from the transverse foramen cranial and caudal to the tumor and ligated, after which discectomies with posterior longitudinal ligament resection were performed in the cephalad and caudad segments. A sagittal osteotomy was created through the vertebral bodies using a high‐speed drill. Next, the entire tumor mass, including the involved vertebral bodies with or without VA and nerve root, was removed en bloc. An operating microscope was used throughout the anterior approach and resection. A titanium cage filled with allograft and/or autograft iliac crest was used for anterior reconstruction. Then anterior plating (Slim‐Loc Anterior Cervical Plate System; DePuy Spine) with screws was fixed to the cephalad and caudal vertebrae.

Results

General Results

Patient characteristics and outcomes are listed in Table 1. There were three men and two women in this case series of mean age at the time of diagnosis and admission 47.2 years (range, 26–67 years). All tumors were located in the subaxial cervical region. All five patients underwent en bloc resection of their cervical spinal tumors via a combined anterior–posterior approach in one stage. The mean operative time was 465 minutes (range, 390–540 minutes) and the mean operative blood loss 1290 mL (range, 1000–1550 mL).

Table 1.

Patient characteristics and surgical data

Case number Age (years) Sex Pathology Location Staging Nerve root sacrifice Vertebral artery sacrifice Blood loss (mL) OR‐Time (min) Margin Complications Follow‐up (months) Status
WBB Enneking
1 28 M Chondrosarcoma C6,7 8–10, A–D, F IB Right C7 Right 1550 540 Marginal UE weakness 27 NED
2 26 F Chondrosarcoma C6,7 4–5,A–C IB None None 1000 420 Wide None 19 NED
3 53 M Chondrosarcoma C5–7 7–9,A–C, F IB Right C6 Right 1400 510 Wide UE pain; UE weakness 22 NED
4 67 M Chordoma C4,5 7–9,A–D, F IB Right C5 Right 1500 466 Marginal Respiratory difficulty, UE weakness 21 NED
5 62 F Chordoma C4 6–7,A–D IB None None 1000 390 Marginal None 16 NED
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F, female; M, male; NED, no evidence of disease; OR, operative time; UE, upper extremity; WBB, Weinstein‐Boriani‐Biagini, including 1–7 zones or 6–12 zones; A–D layers, with or without unilateral vertebral artery (F layer).

Histologic Findings

The excised specimens were sent to the Pathology Departement for histologic analysis and assessment of tumor margins. The pathologic diagnoses were chondrosarcoma in three patients and chordoma in two. Tumor margins were reported as wide, marginal, or intralesional. Two cases (Cases 2 and 3) were reported as having wide margins and three (Cases 1, 4 and 5) as marginal (Table 1).

Complications and Adjuvant Therapy

The VA was sacrificed unilaterally in three patients (Fig. 1) and preserved in two patients (Fig. 2). Nerve roots involved by tumor were sacrificed in three patients who had preoperative upper extremity weakness. There were no intraoperative complications. No cerebrovascular complications or wound infection were observed. Postoperative complications included upper extremity weakness and/or radicular pain in three patients and respiratory difficulties and pneumonia in one patient with a C4 and C5 chordoma (Case 4). This patient recovered completely after 2 weeks’ management with a tracheotomy tube and antibiotics in the intensive care unit. The thrree patients with marginal resection underwent radiotherapy postoperatively, a total dose of 45 Gy being administered in 20 fractions. All patients wore a collar or halo orthosis for 3 months. The average duration of follow‐up was 21 months with a range of 16–27 months. No local recurrence or instrumentation failure was detected on CT or MR imaging during follow‐up.

Figure 1.

Figure 1

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Case 1, (A, B) Preoperative T2 weighted sagittal and axial MR images showing an extensive soft tissue mass with spinal cord compression at C6 and C7. (C, D) Sagittal and axial CT angiograms showing that the right vertebral artery is encased by the mass. (E) Preoperative 3‐D‐printing model showing tumor involving the right vertebral artery and vertebral bodies of C6, 7. (F, G) Schematic diagrams of sagittal en bloc resection via a combined posterior–anterior approach were designed guided by the model. (H) Intraoperative photo: after placement of pedicle screws, laminectomy was performed to ensure a wide posterior margin. (I) Intraoperative photo showing sagittal osteotomies from an anterior direction using a high‐speed drill after ligation of the right vertebral artery. (J) Intraoperative photo after en bloc resection of the tumor showing scarification of the right vertebral artery and C7 nerve root. (K) Intraoperative photo showing ventral reconstruction of the cervical spine. (L) Photo of en bloc excised tumor. (M, N) Postoperative anteroposterior and lateral plain radiographs showing the position of the instruments. (O) Pathological examination resulted in a final diagnosis of chondrosarcoma. HE staining; magnification ×20.

Figure 2.

Figure 2

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Case 2. (A, B) Axial and coronal MR images after the administration of gadolinium contrast showing a tumor centered on the C7 vertebra and extending into the C6 vertebra. (C) Sagittal CT angiogram image showing a soft tissue mass without encasement of the left vertebral artery. (D) Preoperative 3‐D‐printing model showing the relationship between the left vertebral artery and tumor. (E) Intraoperative photo: after placement of pedicle screws, laminectomy and dissection of the nerve roots has been performed to ensure a wide posterior margin. (F) Intraoperative photo showing soft tissue and left vertebral artery at C6‐C7 levels have been fully dissected. (G) Intraoperative photo showing en bloc removal of the tumor has been performed after sagittal osteotomies and discectomies. (H) Intraoperative photo showing ventral view of the dura after en bloc resection of the tumor; the left VA and C6–C8 nerve roots are intact. (I) Photo of en bloc excised tumor. (J) Postoperative anteroposterior plain radiographs demonstrating the position of the instruments. (K) Pathological examination revealed chondrosarcoma HE staining; magnification ×20.

Discussion

Modern advances in spinal tumor staging, imaging, surgical techniques and instrumentation materials have enabled aggressive and radical management of spinal tumors1, 2, 3, 4, 5, 6, 7, 8, 9, 25, 33, 35. En bloc resection of primary spinal tumors offers the following benefits5, 7, 8, 36: (i) avoidance of contamination by tumor cells and correspondingly decreased rates of local recurrence; (ii) the possibility of surgical cure; and (iii) reduced intraoperative blood loss. Although it is technically a challenging procedure, en bloc resection has been well accepted in spinal oncological surgery5, 8, 13, 36.

Indications

Total en bloc spondylectomy has been widely used to treat malignant tumors of the thoracic and lumbar spine. In the TES procedure, the whole vertebra with the entire posterior elements as two separate parts is removed in an en bloc manner7, 8, 33. This technique enables total en bloc resection of one or multiple consecutive vertebrae. However, certain anatomical features of the cervical spine make it unsuitable for TES of cervical spinal tumors. These features include the transverse foramen, VAs and sub‐axial nerve roots, which need to be preserved: segmental arteries and nerve roots must usually be sacrificed bilaterally when TES is performed10, 11, 14. It is reportedly almost impossible to achieve en bloc resection of the cervical spine without sacrificing these vital structures7, 8, 10, 11, 12, 20, 23. Therefore, the indications for en bloc resection in the cervical spine differ from those for the thoracic and lumbar spine. According to systematic reviews, the indications for en bloc resection include: (i) primary malignant or aggressive benign spinal tumors; (ii) solitary lesion without evidence of disseminated disease; and (iii) Enneking grades 3, IA, IB, IIA or IIB; WBB staging: 4–8, 5–9, or 10–3 sectors, A–D layers, with or without unilateral VA (F layer)10, 11, 12, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27.

Management of VAs and Surgical Margins

Technically, en bloc resection of cervical spinal tumors is achieved mainly through a sagittal osteotomy10, 11, 14, 15, 18, 19, 20, 21, 22, 23. The tumor together with the ipsilateral involved segmental VA and nerve roots is removed an en bloc without intralesional resection. Because there is usually unilateral VA and cervical nerve root involvement with eccentric cervical spinal tumors, the potential need for unilateral VA ligation and nerve root sacrifice must be seriously considered10, 11, 14, 15, 18, 19, 20, 21, 22, 23. Although there are no reported instances of serious complications, the risk is high because of the variability of the branches of the VA. Some authors believe that preoperative angiography, balloon occlusion tests and assessment of cerebral blood flow are effective means of evaluating the risk of unilateral VA ligation10, 11, 14, 15, 18, 19, 20, 21, 22, 23. In our case series, we performed preoperative balloon occlusion tests to assess the blood flow of the contralateral VA and detected no vascular malformations or insufficient blood supply.

En bloc resection can be subdivided into “intralesional”, “marginal”, or “wide” categories by gross and histological examination of the operative specimen13, 33. According to the principles of the Enneking oncological surgical classification, the optimal surgical outcome is achievement of a wide margin. In 1999, Fugita et al. reported a case of en bloc resection of chordoma at C5 using a special designed T‐saw through an anterior approach10. They removed the tumor plus the ipsilateral transverse process and VA en bloc. However, because a small amount of residual tumor was detected along the C6 nerve root, strictly speaking this was an intralesional rather than en bloc resection. As stated previously, when en bloc resection is planned, the need to sacrifice the VA and the nerve roots unilaterally must be considered. A combined anterior–posterior surgical approach is preferred to ensure adequate exposure. Molina et al. retrospectively reviewed 16 patients who had undergone en bloc resection of cervical chordomas23. All of their subaxial cases and the majority (57%) of C1, 2 cases were treated via a combined approach. In our case series, all five patients underwent en bloc resection via a combined approach, which is consistent with similar published cases12, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27. With lesions extending into the epidural space, dissection should be performed along the pseudocapsule or the layer of reactive tissue surrounding the tumors, this being defined as marginal resection. In our case series, two cases underwent wide excision and the other three marginal resection.

3‐D Printing Model

It is important to devise a meticulous and individualized surgical plan to achieve a safe resection margin. The central difficulty with this procedure is how to plan precisely preoperatively to achieve an adequate margin. The advent and expanded use of 3‐D printing techniques in the field of medicine have led to a number of innovations, especially through the building of patient‐specific models based on actual imaging data28, 29, 30, 31, 32. This technique provides a reliable reference for surgical planning. A 3‐D printing spinal tumor model provides an accurate representation of the tumor location, size and shape. It is therefore useful in surgical planning and maximizes the possibility of en bloc resection, as well as providing individualized information concerning cervical spinal reconstruction after tumor resection. Giovinco et al. have reported that using 3‐D printing models for preoperative planning of Charcot foot reconstruction assists selection of optimal incision placement, instrumentation, osteotomy and joint resection, and placement of internal and external fixation devices32. Similarly, Guppy et al. used individually reconstructed models to design the surgical approach to upper cervical spine lesions and successfully performed en bloc resections22. Currently, the use of 3‐D printing models in medicine assists not only surgical planning, but also practicing and training. Waran et al. reported that 3‐D printing models are useful for training neurosurgeons30, providing an important supplement to and improving their training. In addition, these models can be used to help patients with cervical spinal tumors and their families understand their tumors, condition, and surgical goals and risks.

Reconstruction

Circumferential reconstruction is mandatory because multilevel en bloc resection results in significant instability. The strategy for reconstruction involves anterior and posterior fixation. Anterior stabilization can be achieved using a titanium cage, plate, autograft, or allograft14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27. Posterior stabilization may be with rod and pedicle and/or later mass screws. We recommend instrumented fixation at least one level above and below the affected vertebra(e); this, not only maintains spinal stability, but also minimizes the effect on cervical function. However, reconstruction of the cervical spine after en bloc resection of tumors in the high cervical region may be extremely difficult. The unique anatomy of this region allows for a variety of reconstruction techniques. To date, no consensus has been reached as to what form of reconstruction is optimal, with the exception of posterior occipitocervical fusion.

Complications

En bloc resection of cervical spinal tumors is feasible, however, complex complications can occur and have adverse effects on outcomes5, 12, 13, 14, 20, 21, 22, 23, 25. Complications of en bloc resection of cervical spinal tumors include postoperative hematoma, neurological defects, cerebrospinal fluid leaks, dysphagia, Horner syndrome, esophageal fistula, recurrent laryngeal nerve palsy, diaphragm paralysis and wound infection12, 14, 18, 19, 20, 22, 23. In our series, three patients had muscle weakness in their upper extremities because their nerve roots had been sacrificed unilaterally. One patient had respiratory difficulties and pneumonia and required 2 weeks of ventilatory support.

There have been only a few reported cases of en bloc resection of primary malignant tumor in the upper cervical spine; however, the surgeons involved believe that this procedure is feasible and improves survival rates20, 21, 22, 23, 24. There is evidence that en bloc resection in the upper cervical spine is associated with higher rates of complication and contaminated margins than with the subaxial cervical spine14, 15, 18, 20, 21, 22, 23, 24. Surgeons must weigh the benefits against the risk of high rates of complications and unfavorable outcomes when tumors are located in the upper cervical spine. Intralesional resection with less destructive approaches is considered the management of choice for atlantoaxial tumors23, 35, 37, 38, 39, 40, 41. Although we have no experience with multilevel en bloc resection of tumors in the upper cervical spine, our study has shown that en bloc resection of primary malignant tumors in the subaxial cervical spine is feasible provided there is a detailed surgical plan and the procedure is performed in specialized centers.

Limitations

The present study has some limitations. First, the small number of patients, a consequence because of the relative rarity of cervical spinal primary malignant tumors and the limited surgical indications, mean that the level of evidence is low. Second, en bloc resection of cervical spinal tumors involves many difficulties, serious complications and long operation times. These disadvantages result in a lengthy learning curve for surgeons and limit its application in clinics. Third, there were no high cervical spinal tumors in this study. Tumor resection and reconstruction after tumor excision in the upper cervical spine is more challenging. In addition, our follow‐up is too short to provide data concerning long‐term tumor control.

Conclusions

En bloc resection via sagittal osteotomy is the most effective means of managing cervical spinal tumors. En bloc resection of eccentric lesions in the subaxial cervical spine with preservation of the VA and nerve roots on one side is feasible. The use of preoperative 3‐D printed models enables better anatomical understanding of the lesion and can contribute to planning of the procedure. Given the surgical complexity, we recommend that this surgery be conducted by an experienced team with multidisciplinary cooperation and meticulous preoperative planning, these being the key points for success.

Disclosure: The authors have no personal financial or institutional interest in any of the drugs, materials, or devices described in this article.

References

  • 1. Boriani S, De Iure F, Bandiera S, et al Chondrosarcoma of the mobile spine: report on 22 cases. Spine (Phila Pa 1976), 2000, 25: 804–812. [DOI] [PubMed] [Google Scholar]
  • 2. Boriani S, Bandiera S, Biagini R, et al Chordoma of the mobile spine: fifty years of experience. Spine (Phila Pa 1976), 2006, 31: 493–503. [DOI] [PubMed] [Google Scholar]
  • 3. Boriani S, Bandiera S, Casadei R, et al Giant cell tumor of the mobile spine. A review of 49 cases. Spine (Phila Pa 1976), 2012, 37: E37–E45. [DOI] [PubMed] [Google Scholar]
  • 4. Stacchiotti S, Casali PG, Lo Vullo S, et al Chordoma of the mobile spine and sacrum: a retrospective analysis of a series of patients surgically treated at two referral centers. Ann Surg Oncol, 2010, 17: 211–219. [DOI] [PubMed] [Google Scholar]
  • 5. Amendola L, Cappuccio M, De Iure F, et al En bloc resections for primary spinal tumors in 20 years of experience: effectiveness and safety. Spine J, 2014, 14: 2608–2617. [DOI] [PubMed] [Google Scholar]
  • 6. Kato S, Murakami H, Demura S, et al More than 10‐year follow‐up after total en bloc spondylectomy for spinal tumors. Ann Surg Oncol, 2014, 21: 1330–1336. [DOI] [PubMed] [Google Scholar]
  • 7. Tomita K, Kawahara N, Baba H, Tsuchiya H, Fujita T, Toribatake Y. Total en bloc spondylectomy. A new surgical technique for primary malignant vertebral tumors. Spine (Phila Pa 1976), 1997, 22: 324–333. [DOI] [PubMed] [Google Scholar]
  • 8. Kawahara N, Tomita K, Murakami H, Demura S. Total en bloc spondylectomy for spinal tumors: surgical techniques and related basic background. Orthop Clin North Am, 2009, 40: 47–63. [DOI] [PubMed] [Google Scholar]
  • 9. Luzzati AD, Shah SP, Gagliano FS, Perrucchini GG, Fontanella W, Alloisio M. Four‐ and five‐ level en bloc spondylectomy for malignant spinal tumors. Spine (Phila Pa 1976), 2014, 39: E129–139. [DOI] [PubMed] [Google Scholar]
  • 10. Fujita T, Kawahara N, Matsumoto T, Tomita K. Chordoma in the cervical spine managed with en bloc excision. Spine (Phila Pa 1976), 1999, 24: 1848–1851. [DOI] [PubMed] [Google Scholar]
  • 11. Cohen ZR, Fourney DR, Marco RA, Rhines LD, Gokaslan ZL. Total cervical spondylectomy for primary osteogenic sarcoma. Case report and description of operative technique. J Neurosurg, 2002, 97 (3 Suppl.): 386–392. [DOI] [PubMed] [Google Scholar]
  • 12. Kaloostian PE, Gokaslan ZL. Surgical management of primary tumors of the cervical spine: surgical considerations and avoidance of complications. Neurol Res, 2014, 36: 557–565. [DOI] [PubMed] [Google Scholar]
  • 13. Yamazaki T, McLoughlin GS, Patel S, Rhines LD, Fourney DR. Feasibility and safety of en bloc resection for primary spine tumors: a systematic review by the Spine Oncology Study Group. Spine (Phila Pa 1976), 2009, 34 (22 Suppl.): S31–S38. [DOI] [PubMed] [Google Scholar]
  • 14. Rhines LD, Fourney DR, Siadati A, Suk I, Gokaslan ZL. En bloc resection of multilevel cervical chordoma with C‐2 involvement. Case report and description of operative technique. J Neurosurg Spine, 2005, 2: 199–205. [DOI] [PubMed] [Google Scholar]
  • 15. Bailey CS, Fisher CG, Boyd MC, Dvorak MF. En bloc marginal excision of a multilevel cervical chordoma. Case report. J Neurosurg Spine, 2006, 4: 409–414. [DOI] [PubMed] [Google Scholar]
  • 16. Currier BL, Papagelopoulos PJ, Krauss WE, Unni KK, Yaszemski MJ. Total en bloc spondylectomy of C5 vertebra for chordoma. Spine (Phila Pa 1976), 2007, 32: E294–E299. [DOI] [PubMed] [Google Scholar]
  • 17. Leitner Y, Shabat S, Boriani L, Boriani S. En bloc resection of a C4 chordoma: surgical technique. Eur Spine J, 2007, 16: 2238–2242. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 18. Cloyd JM, Chou D, Deviren V, Ames CP. En bloc resection of primary tumors of the cervical spine: report of two cases and systematic review of the literature. Spine J, 2009, 9: 928–935. [DOI] [PubMed] [Google Scholar]
  • 19. Chou D, Acosta F Jr, Cloyd JM, Ames CP. Parasagittal osteotomy for en bloc resection of multilevel cervical chordomas. J Neurosurg Spine, 2009, 10: 397–403. [DOI] [PubMed] [Google Scholar]
  • 20. Hsieh PC, Gallia GL, Sciubba DM, et al En bloc excisions of chordomas in the cervical spine: review of five consecutive cases with more than 4‐year follow‐up. Spine (Phila Pa 1976), 2011, 36: E1581–E1587. [DOI] [PubMed] [Google Scholar]
  • 21. Vassal F, Dumas B, Nuti C. Margin‐free, en bloc resection of a multilevel cervical chordoma with C‐2 involvement: case report with 2‐year follow‐up and description of operative technique. Br J Neurosurg, 2012, 26: 929–931. [DOI] [PubMed] [Google Scholar]
  • 22. Guppy KH, Chakrabarti I, Isaacs RS, Jun JH. En bloc resection of a multilevel high‐cervical chordoma involving C‐2: new operative modalities: technical note. J Neurosurg Spine, 2013, 19: 232–242. [DOI] [PubMed] [Google Scholar]
  • 23. Molina CA, Ames CP, Chou D, et al Outcomes following attempted en bloc resection of cervical chordomas in the C‐1 and C‐2 region versus the subaxial region: a multiinstitutional experience. J Neurosurg Spine, 2014, 21: 348–356. [DOI] [PubMed] [Google Scholar]
  • 24. Ortega‐Porcayo LA, Cabrera‐Aldana EE, Arriada‐Mendicoa N, Gómez‐Amador JL, Granados‐García M, Barges‐Coll J. Operative technique for en bloc resection of upper cervical chordomas: extended transoral transmandibular approach and multilevel reconstruction. Asian Spine J, 2014, 8: 820–826. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 25. Mattei TA, Mendel E. En bloc resection of primary malignant bone tumors of the cervical spine. Acta Neurochir(Wien), 2014, 156: 2159–2164. [DOI] [PubMed] [Google Scholar]
  • 26. Štulík J, Barna M, Vyskočil T, Nesnídal P, Kryl J, Klézl Z. Total en bloc spondylectomy of C3: a new surgical technique and literature review. Acta Chir Orthop Traumatol Cech, 2015, 82: 261–267. [PubMed] [Google Scholar]
  • 27. Lee CH, Min WK. Cervical subtotal en‐bloc spondylectomy of C6 mesenchymal chondrosarcoma. Eur Spine J, 2015. . [Epub ahead of print]. [DOI] [PubMed] [Google Scholar]
  • 28. Rengier F, Mehndiratta A, von Tengg‐Kobligk H, et al 3D printing based on imaging data: review of medical applications. Int J Comput Assist Radiol Surg, 2010, 5: 335–341. [DOI] [PubMed] [Google Scholar]
  • 29. Rankin TM, Giovinco NA, Cucher DJ, Watts G, Hurwitz B, Armstrong DG. Three‐dimensional printing surgical instruments: are we there yet? J Surg Res, 2014, 189: 193–197. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 30. Waran V, Narayanan V, Karuppiah R, Owen SL, Aziz T. Utility of multimaterial 3D printers in creating models with pathological entities to enhance the training experience of neurosurgeons. J Neurosurg, 2014, 120: 489–492. [DOI] [PubMed] [Google Scholar]
  • 31. Wu AM, ZX S, JS W, et al The accuracy of a method for printing three‐dimensional spinal models. PLoS One, 2015, 10: e0124291. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 32. Giovinco NA, Dunn SP, Dowling L, et al A novel combination of printed 3‐dimensional anatomic templates and computer‐assisted surgical simulation for virtual preoperative planning in Charcot foot reconstruction. J Foot Ankle Surg, 2012, 51: 387–393. [DOI] [PubMed] [Google Scholar]
  • 33. Boriani S, Weinstein JN, Biagini R. Primary bone tumors of the spine. Terminology and surgical staging. Spine (Phila Pa 1976), 1997, 22: 1036–1044. [DOI] [PubMed] [Google Scholar]
  • 34. Enneking WF. A system of staging musculoskeletal neoplasms. Clin Orthop Relat Res, 1986, 204: 9–24. [PubMed] [Google Scholar]
  • 35. Suchomel P, Buchvald P, Barsa P, et al Single‐stage total C‐2 intralesional spondylectomy for chordoma with three‐column reconstruction. Technical note. J Neurosurg Spine, 2007, 6: 611–618. [DOI] [PubMed] [Google Scholar]
  • 36. Cloyd JM, Acosta FL Jr, Polley MY, Ames CP. En bloc resection for primary and metastatic tumors of the spine: a systematic review of the literature. Neurosurgery, 2010, 67: 435–444. [DOI] [PubMed] [Google Scholar]
  • 37. Yang X, Wu Z, Xiao J, et al Sequentially staged resection and 2‐column reconstruction for C2 tumors through a combined anterior retropharyngeal‐posterior approach: surgical technique and results in 11 patients. Neurosurgery, 2011, 69 (2 Suppl Operative): ons184–ons193 ; discussion 193–14. [DOI] [PubMed] [Google Scholar]
  • 38. Yang X, Huang W, Xiao J, et al Combined pre‐ and retrovascular extraoral approach for tumors at lateral mass of the atlas. Spine (Phila Pa 1976), 2011, 36: 129–136. [DOI] [PubMed] [Google Scholar]
  • 39. Štulík J, Kozák J, Šebesta P, Vyskočil T, Kryl J, Klezl Z. Total spondylectomy of C2: report of three cases and review of the literature. J Spinal Disord Tech, 2010, 23: e53–e58. [DOI] [PubMed] [Google Scholar]
  • 40. Menezes AH, Ahmed R. Primary atlantoaxial bone tumors in children: management strategies and long‐term follow‐up. J Neurosurg Pediatr, 2014, 13: 260–272. [DOI] [PubMed] [Google Scholar]
  • 41. Barrenechea IJ, Perin NI, Triana A, Lesser J, Costantino P, Sen C. Surgical management of chordomas of the cervical spine. J Neurosurg Spine, 2007, 6: 398–406. [DOI] [PubMed] [Google Scholar]

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