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Technical Notes

Osamu Ikeda, MD, PhD,1 Norihiko Minami, MD,1 Masashi Yamazaki, MD, PhD,2 Masao Koda, MD, PhD,3 Tatsuo Morinaga, MD, PhD1

1Department of Orthopedic Surgery, Kashiwa City Kashiwa Hospital, Kashiwa, Chiba, Japan,
2Department of Orthopedic Surgery, Tsukuba University, Tsukuba, Ibaraki, Japan
3Department of Orthopedic Surgery, Chiba University Graduate School of Medicine, Chiba, Chiba, Japan

Address correspondence to:
                Osamu Ikeda, MD, PhD
                Department of Orthopedic Surgery
                Kashiwa City Kashiwa Hospital
                1-3 Fuse, Kashiwa, Chiba 277-0825
                Japan
                Tel: +81-4-7134-2000
                Fax: +81-4-7134-2001
                E-mail:  HYPERLINK "mailto:ikeda@kashiwacity-hp.or.jp" ikeda@kashiwacity-hp.or.jp


Conflicts of Interest and Source of Funding:
The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this manuscript.
The authors received no outside financial support for the procedure described in this manuscript.
Informed consent:
The patient and their families were informed that data from the case would be submitted for publication, and gave their consent.
ABSTRACT
Background: Severe cervical kyphosis produces persistent axial neck pain and difficulty with forward gaze, a major source of these symptoms is neck extensor muscles placed in an overstretched condition. The recent trend of surgical strategies for cervical kyphosis is towards posterior instrumentation because of its advantages in powerful correction and rigid fixation. From the standpoint of treating neck extensor impairment, surgery that is less invasive to the nuchal muscles should be given priority. Because of uncertainty in correction and fixation forces, anterior surgery is rarely given challenge to cervical kyphosis as severe as 50� or more. The purpose of this report is to describe our anterior surgical technique to address severe cervical kyphosis and present a case of cervical kyphosis of 64� at C2�C7 in a 63-year-old woman who underwent successful correction using this technique.
Methods: We repeated spreading and interbody bone grafting at each segment of from C3 through C7 without any corpectomies. We placed anchor screws with a plate into 5 vertebral bodies, and augmented this posteriorly by in situ fastening the spinous processes with a cable under a minimally invasive exposure where nuchal muscles were preserved.
Results: We achieved a correction of up to 8� kyphosis using this technique. Bony fusion was completed without any loss of correction at 2 years follow-up. Our patient was released from persistent nuchal pain and became able to hold her head in an upright position.
Conclusions: Anterior surgery addresses severe kyphosis, and has the advantage of posterior surgery in regard to sparing the neck extensor muscles. The presented technique could be considered as a surgical option for this pathology.

Keywords: Cervical kyphosis, Anterior cervical discectomy and fusion, Combined anterior and posterior fusion, Neck extensor muscle





















INTRODUCTION
Cervical kyphosis (CK) produces, in cases where it is as severe as 50� or more, persistent axial neck pain and difficulty with forward gaze as well as neural symptoms. Axial neck pain, which is one of the main targets of treatment, is thought to arise from neck extensor muscles placed in an overstretched condition in addition to degenerated discs [1]. Furthermore, difficulty with forward gaze is from absolute or relative weakness of the neck extensor muscles [1, 2]. From the standpoint of treating the impairment of neck extensor muscles, surgery that is less invasive to these muscles is preferable [1]. Posterior instrumentation surgery and posterior instrumentation with anterior bone grafting have become more prevalent since remarkable advances in spinal instrumentation have recently allowed strong correction and rigid fixation [2�6]. However, posterior surgery presents a serious problem because of potential invasion of the neck extensors [7, 8]. This problem raises the question of whether surgeons can perform sufficient correction and fixation with anterior surgery for CK as severe as 50� or more. In this report, we describe our anterior correction surgery with some ideas to resolve its potential problems, and present a case of severe CK in a patient who achieved successful correction and fixation by means of this technique.

MATERIALS AND METHODS
The patient and her family were informed that data from the case would be submitted for publication and gave their consent.
History and examination
A 63-year-old woman developed nuchal pain and stiffness about 30 years ago. She first visited a hospital 10 years ago and was diagnosed with cervical spondylosis. Head dropping and CK found in roentgenography began to develop 2 years ago. Axial neck pain grew more serious 6 months ago such that she could not maintain an upright head position during standing or sitting; she eventually needed to support her head with her hand while eating a quick meal. Neurological tests indicated a slight loss in grasping power that was 12 kg on her left and also mild hypoalgesia that existed in the area from her C5 to C8 dermatomes. Her Japanese Orthopaedic Association (JOA) score was 14 of 17 [9]. Roentgenograms showed kyphosis of 38� at C2�C7 two years before surgery (Figure 1A), which progressed to 44� in the next 18 months (Figure 1B), and rapidly progressed to 64( in the subsequent 6 months (Figure 1C). There was no evidence of instability between C2 and C3, whereas C2 showed anterolisthesis. A computed tomographic (CT) myelogram showed that vertebral discs bulged, and retrovertebral bony spurs were formed in 4 segments from C3-4 through C6-7 (Figure 1D�F), both of which participated in flattening the spinal cord in concert with anterior shifting of the spinal cord as a result of kyphosis. Blood tests excluded endocrinologic or metabolic disease. Interestingly, blood tests showed creatine phosphokinase (CPK), one of the leaking enzymes of muscle origin, gradually increasing along with CK progression (Figure 2). However, electromyography provided no evidence of a myopathy. She was diagnosed with rapidly progressing degenerative CK and was scheduled for surgical treatment.
Surgical procedure
Beginning with a discectomy of 4 segments between C3-4 and C6-7 via an anterior approach, the retrovertebral bony spurs were then removed through the interbody space. An angled small curette was used to remove bony spurs without excising the posterior longitudinal ligament (PLL) (Figure 3A). Special attention was paid to preserve the subchondral bone of each end plate and PLL because an excessively damaged end plate can result in sinking of the grafted bone (Figure 3B). Moreover, the PLL plays an important role in the ligamentotaxis mechanism by which kyphosis can be easily corrected by spreading the intervertebral body space, and grafted bone can be stabilized through the fusion process (Figure 3C). A microscope is helpful for these procedures in the small field. After completion of decompression, autologous tricortical iliac bone, 5 to 8 mm in standard height, was introduced into the intervertebral body, spread as much as possible by using a spreader. Kyphosis was safely and gradually corrected by repeating the process of spreading and interbody bone grafting segment by segment. It was better to begin bone grafting at the segment marking the kyphosis apex because it required the biggest spacer, and space became more restricted in the later segments. We recommend obtaining a roentgenogram after placing the trial spacer to check how much deformity is corrected and to determine the size of graft bone before grafting (Figure 3D, E). Where the correction is not sufficient, the Luschka joint may need to be removed. After bone grafting in all 4 segments (Figure 3F), the anterior plate was placed. The stability of the anterior plating was ensured by using 10 anchor screws: 5 vertebral bodies receiving 2 each [10]. Nevertheless, anterior plating with multiple segmental fusions was not considered sufficiently durable for rigid kyphosis [11]. We therefore augmented the plating posteriorly by fastening the spinous processes with a plastic cable from C3 to C7 to reduce postoperative correction loss. Our experience has proven this supplemental procedure to be indispensable to prevent correction loss. We limited the exposure of the spinous processes as much as possible without taking off the erector spinae muscles attaching them to the lamina (Figure 3G). Plastic or polyethylene cable has advantages for fastening the spinous processes because of its ease of handling compared with conventional wire cable. This posterior augmentation is aimed at preventing the interspinous interval from opening, not for correcting the deformity that has already been corrected through the anterior surgery. Bone chips were grafted on the base of spinous processes at C2-3 and C4-5 in this case (Figure 4A�C).

RESULTS 
Clinical outcome
A Philadelphia collar was applied to the patient, who was allowed to walk the day after surgery. She was allowed free neck motion 3 months after surgery. Nuchal pain dramatically diminished, and she regained the ability to hold her head in an upright position, resulting in major improvements in her activities of daily living. Grasping power was improved to 21 kg on her left. Her JOA score increased to 16 of 17. She felt mild difficulty in swallowing; however, this difficulty decreased in 3 months. No other complications were reported. Additionally, we found that the preoperative rise in CPK levels fell immediately after surgery and that CPK increased again with the muscle exercises starting at 2 months after surgery, returning to normal during the 2 years after surgery (Figure 2).
Radiological outcome
Postoperative CT showed that the CK was impressively corrected to 8� kyphosis and bony spurs were thoroughly excised (Figure 4A�C). The correction angle achieved by this surgery was notably 56�. A roentgenogram at the two-year follow-up showed that bony union of all 4 segments was completed in the state of 8( kyphosis. This implies that no correction loss had occurred through the bone uniting process (Figure 4E).

DISCUSSION
Our technique involves 4 segments of anterior cervical discectomy and fusion (ACDF) supplemented with minimally invasive posterior tension bands. To our knowledge, there have been no reports explaining the details of this technique in patients with severe kyphosis. Using this technique, we succeeded in obtaining a large CK correction from 64� preoperatively to 8� postoperatively without any postoperative correction loss. Additionally, there was dramatic functional recovery of forward gaze with complete relief of the patient�s nuchal pain. Furthermore, our findings that CPK fluctuated along with CK progression and muscle loading suggested a close relationship between CK and the neck muscles.
To summarize the strong points of the present surgery: it safely corrects the deformity by repeating spread and bone graft in each segment, with little risk of graft dislodgement and sinking obtained by dispersing the axial load onto each segment; an iliac autograft enables an earlier union; many anchor screws can be placed because all the vertebral bodies are preserved; and supplementary interspinous tension bands under minimally invasive exposure preserve neck extensors. We think that all of these aspects can contribute to good outcomes both radiologically and symptomatically. This technique does not necessitate a halo vest, even when performing a large correction of 50� or more. Although posterior tension bands need to expose spinous processes, the less invasive nature of this procedure was shown by the fact that the preoperative rise in CPK levels did not worsen postoperatively but immediately fell on the day after surgery. On the other hand, limitations of this surgery include that it is contraindicated in patients who have posterior ankylosis, such as postlaminectomy kyphosis [12�14]. This is because we can perform neither spinous process fastening nor posterior release under minimal exposure. Additionally, extending the fixation area craniocaudally is restricted in this surgery. We must also be aware that anterior surgery has the potential for injury to organs, including the bronchus and esophagus.
Conventionally, anterior surgery for severe CK requires long fusion of either 4 or 5 segments with a strut bone graft following consecutive corpectomies; however, this can cause problems during the surgery and through the bone uniting process. For instance, fracture of the vertebral body receiving the graft and sinking of grafted bone leads to correction loss because the axial load impinges on only two sites [15, 16]. It is difficult to prevent correction loss and implant failure when acquiring a large correction even if anterior plating and a halo vest are used together [1, 14, 17�19]. Multilevel ACDF has recently been indicated for spondylosis, and differing from corpectomies, it has been reported to contribute to biomechanical stability [10, 20]. However, to our knowledge, there have been no reports where multilevel ACDF surgery without posterior instrumentation has been challenged by CK as severe as 50� or more that required a large correction and rigid fixation because of the unreliability of the correcting and fixating force. In the present report, we are the first to demonstrate that ACDF has excellent performance in the case of severe CK.
Posterior instrumentation surgery and posterior instrumentation with anterior bone grafting have become more prevalent since remarkable advances in spinal instrumentation have recently allowed strong correction and rigid fixation [2�6]; however, posterior surgery presents a serious problem because of potential invasion of the neck extensors. Indeed, several authors have indicated that preservation of neck extensor muscles works effectively for preventing postoperative axial neck pain [7, 8]. Accordingly, we can say that posterior surgery is not necessarily the best treatment for CK for this reason.
We interpret CK as a type of neck extensor impairment. Symptoms involved in severe CK are persistent axial neck pain and difficulty with forward gaze as well as neural symptoms, and a major source of the former is neck extensor muscles placed in an overstretched condition. Therefore, we argue the preference for anterior surgery, which is less invasive of neck extensor muscles, over posterior surgery in this report. Although our surgery cannot resolve all the morbidity associated with CK arising from various origins, it could be considered as a surgical option.

CONCLUSIONS
We report successful surgery for a patient with severe CK using 4 segments of ACDF supplemented with minimally invasive posterior tension bands. This technique appears safe and provides for sufficient correction and anterior decompression, with very little postoperative correction loss. We showed that anterior surgery can address kyphosis as severe as 50� or more and also retains the advantage of posterior surgery with regard to sparing the neck extensor muscles.


Disclosure
The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this manuscript.
The authors received no outside financial support for the procedure described in this manuscript. 
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FIGURE LEGENDS
Fig. 1: Progressive cervical kyphosis before surgery
Roentgenogram showing progressive cervical kyphosis: 38� at C2�C7 two years before surgery (A), which progressed to 44� in the next 18 months (B), and made rapid progress to 64( in the subsequent 6 months (C). An axial CT myelographic view, C4�C5 (D), C5�C6 (E) and C6�C7 (F), shows the spinal cord flattened by a vigorous retrovertebral bony spur in concert with anterior shifting of the cord

Fig. 2: Correlation between creatine phosphokinase (CPK) and the CK angle 
Graph showing time-course changes of creatine phosphokinase (CPK) and the CK angle. These two parameters appear correlated except for a temporary rise of CPK during the muscle exercises after surgery

Fig. 3: Photographs and roentgenogram during surgery
Intraoperative photographs showing the retrovertebral bony spurs removed using an angled curette (A), preserved subchondral bone (B, arrow), the intervertebral body space widened with a spreader (C), and the trial spacer being inserted into interbody space (D). Intraoperative roentgenogram (E) taken for checking the correction angle and cervical alignment. Intraoperative photographs showing tricortical iliac bone (F, arrowheads) between vertebral bodies (F, arrows) in the anterior procedure, and spinous processes (G, arrows) fastened together with a plastic suture (G, arrowheads) in the posterior procedure. Note that there is minimal detachment of the neck extensors from the lamina and spinous processes. The cranial direction is rightmost in all photographs

Fig. 4: Images after surgery 
Reconstructed sagittal (A) and axial CT views at C4�C5 (B), C5�C6 (C), and C6�C7 (D) 1 week after surgery show that the deformity was impressively corrected and bony spurs were thoroughly excised. Arrowheads indicate bone chips grafted onto the base of the spinous processes. Roentgenogram obtained 2 years after surgery (E) showing completed bone union with no correction loss











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