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 Table of Contents  
CASE REPORTS
Year : 2022  |  Volume : 5  |  Issue : 2  |  Page : 231-234

Iatrogenic fracture in a patient of ankylosing spondylitis planned for surgical correction of chin-on-chest deformity: A case report and review of literature


Department of Spine Surgery, Kothari Medical Centre, Kolkata, India

Date of Submission11-Jul-2021
Date of Acceptance12-Aug-2021
Date of Web Publication08-Jun-2022

Correspondence Address:
Kushal Gohil
Department of Spine Surgery, Kothari Medical Centre, 8/3 Alipore Road, Alipore, Kolkata 700027, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/isj.isj_66_21

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  Abstract 

Ankylosing spondylitis (AS) is a progressive systemic chronic inflammatory rheumatic disease that causes arthritis of spine and sacroiliac joints and finally results in contracture or bony fusion of these joints. The ankylosed spine has increased propensity to fracture due to rigidity, long lever arms, and osteoporosis. However, iatrogenic fracture occurring in AS patients with chin-on-chest deformity during positioning has not been reported. A 46-year-old male, known case of AS, presented with progressive deformity of neck for the last 6 months due to C5 fracture with C4/5 dislocation. He had chin-on-chest deformity with chin-brow vertical angle of 106o with spastic tetraparesis (mJOA score 6, Nurick grade-3). The patient was put on sustained halo-gravity traction by gradually increasing the weight up to 30 lbs for 4 weeks and then was planned for deformity correction. After intubation and application of same traction, we found anterior wedge opening and fracture through C6/7 disc space. His neurology was found to be same as in the preoperative state on neural monitoring and wake up test. Front and back instrumented fusion was done with anterior bone grafting. Postoperatively, he was maintained on halo-vest immobilization for three months. By the end of first year, the fracture had united well and he was mobilizing well with mJOA score 13 and Nurick grade-1. AS patients are at higher risk of fracture and need utmost care while positioning during surgery. Traction or any corrective maneuver must be done carefully.

Keywords: Ankylosing spondylitis, chin-on-chest deformity, iatrogenic spinal fracture


How to cite this article:
Basu S, Gohil K, Gupta A, Gowtham JR, Manikanta D, Bandagi G, Biswas A. Iatrogenic fracture in a patient of ankylosing spondylitis planned for surgical correction of chin-on-chest deformity: A case report and review of literature. Indian Spine J 2022;5:231-4

How to cite this URL:
Basu S, Gohil K, Gupta A, Gowtham JR, Manikanta D, Bandagi G, Biswas A. Iatrogenic fracture in a patient of ankylosing spondylitis planned for surgical correction of chin-on-chest deformity: A case report and review of literature. Indian Spine J [serial online] 2022 [cited 2022 Jul 1];5:231-4. Available from: https://www.isjonline.com/text.asp?2022/5/2/231/346973




  Introduction Top


Ankylosing spondylitis (AS) is a progressive systemic chronic inflammatory rheumatic disease that causes arthritis of spine and sacroiliac joints and finally results in contracture or bony fusion of these joints. In severely affected patients, the entire spine, including the pelvis, becomes fixed in a flexed position.[1] In severe cases, fixed cervical kyphotic deformity may develop, which may affect horizontal gaze function, impede activities of daily living (such as swallowing), and cause disabling pain.[2] Corrective surgery is advocated in such cases. The ankylosed spine has increased propensity to fracture due to rigidity, long lever arms, and osteoporosis.[3],[4]

In this case report, we present our experience with the correction of chin-on-chest deformity in a patient with AS, who developed iatrogenic fracture during positioning and the final outcome.


  Case Report Top


A 46-year-old male with longstanding AS presented with progressive flexion deformity of neck for 6 months after history of fall. He had weakness of all 4 limbs with urinary urge incontinence and gait instability for the past three months. He had difficulty in gazing upwards and opening his mouth.

On examination, he had severe kyphotic deformity of cervical spine with spastic tetraparesis and motor power grade 3–4/5 in all four limbs [Figure 1]A. All deep tendon reflexes were brisk, and plantars were extensor with mJOA score 6 and Nurick grade-3.
Figure 1: (A) The patient presented with severe kyphotic deformity of cervical spine with inability to keep horizontal gaze and the chin-brow vertical angle (CBVA) of 106o. (B-D) X-ray and CT scan findings at 6 months post-trauma showing local kyphosis (angle 70°) with sclerosis and wedging of C4 body

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Imaging demonstrated a severe sagittal cervical deformity with local kyphotic angle of 70o and fracture of C5 with C4/5 dislocation [Figure 1B-D].

The patient was put on sustained halo-gravity traction by gradually increasing the weight up to 30 lbs. At the end of 4 weeks, there was significant soft tissue stretching without much opening of deformity anteriorly as evident by the serial portable radiographs [Figure 2]. He was then planned for deformity correction with T1 pedicle subtraction osteotomy and posterior fixation.
Figure 2: (A) Halo gravity traction applied in ward showing at least four finger breadths anterior opening of neck. (B) Portable X-ray showing no change in the cervical kyphosis even after sustained halo traction for 4 weeks

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Surgery details

After fiber optic-assisted nasal intubation, the same traction (30 lbs) was applied in the supine position. Thereafter, we encountered abnormal anterior opening, and fluoroscopy showed abnormal anterior wedge opening and fracture through C6/7 disc space. Neural monitoring did not show any loss of signals. The wake-up test was performed and the patient’s neurology was found to be same as in the preoperative state. Decision was made to do an in-situ front back fixation and fusion. Through anterior right-sided approach, a contoured anterior cervical plate was fixed across C6/7 and the void was filled with iliac crest bone graft [Figure 3]. The patient was then flipped over and correction of deformity was noted after positioning. Instrumented posterior spinal fusion was done from C2 to T5 after an initial short segment C6 C7 T1 fixation.
Figure 3: (A) Abnormal anterior opening (eight finger breadths) seen after he was put on same traction of 30 lbs after intubation. (B) The C-arm image showing wedge opening anteriorly at C6/7 disc space. (C) Fracture fixed anteriorly with contoured plate and iliac crest graft

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Postoperative protocol

His postoperative recovery was uneventful. He was maintained on halo-vest immobilization and was mobilized out of bed on 3rd postoperative day. At three months of follow-up, halo-vest removal done and Sternal-Occipital-Mandibular Immobilizer (SOMI) brace applied [Figure 4]A and B. He did well at 1-year follow-up [Figure 4]C and D and was mobilizing well with brace with mJOA score 13 and Nurick grade-1.
Figure 4: (A) Immediate postoperative X-ray showing satisfactory position of implants and good correction of deformity. (B) Halo-vest immobilization done postoperatively for 3 months. (C) One-year follow-up with significant improvement in neurology and forward gaze (CBVA of 38o). (D) X-rays at 1-year follow-up showing satisfactory position of implants and good correction of deformity with fusion and integration of graft at C6/7

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  Discussion Top


Intra-operative iatrogenic fracture occurring during spine surgery is a rare complication. In the present case, there was an unintentional open wedge fracture which occurred at C6/7 when the patient was being positioned after intubation and application of traction. We found only three such published case reports of intra-operative iatrogenic fractures in AS patients.[3],[4] Ruf et al.[3] reported two such cases, the first case being a 39-year-old male who was operated on for cervical stenosis due to C1/2 instability and developed acute quadriplegia due to C6/7 fracture/dislocation noticed postoperatively. In the second case, a 55-year-old male, who was undergoing thoracic deformity correction, developed iatrogenic fracture at C6/7 while being under anesthesia. Both these cases required additional fixation. Maruiwa et al.[4] reported a case of 66-year-old woman with chin-on-chest deformity caused by upper cervical (C0-2) kyphosis associated with AS. Deformity correction with C0-5 posterior fixation was done. There was C3/4 open wedge fracture after the lordotic rods were captured, and the fixation was revised with extension of fusion to T3 with iliac bone grafting.

In the present case, the fixed fragment from C0-6 formed longer lever arms, which probably added stress on the C6/7 disc level and resulted in fracture at that level. We did not anticipate that the same cervical traction of 30 lbs which was applied in ward with added weight of patient’s head could cause additional stress and tension and cause a fracture under the effect of muscle relaxation and general anesthesia. The cervico-thoracic junction seems most vulnerable as it is a site of stress concentration and zone of transition between lordotic, flexible cervical spine and kyphotic, rigid thoracic spine.[5]

First, we did a circumferential fusion at the fracture site (anterior and posterior fixation) to decrease risk of neurological injury due to instability and fracture translation. Secondly, to improve the stability, long posterior fixation from C2-T5 was done. Thirdly, we immobilized with halo-vest postoperatively till signs of bony fusion appeared.

Despite the surgical complication, the clinical outcome of this patient is really inspiring. He not only got kyphosis correction and improved frontal gaze but also improved his neurology and satisfaction about his appearance. However, the risk of spinal cord injury, intraoperative sagittal translation, and post-operative instrumentation failure should always be considered.


  Conclusion Top


AS patients are at higher risk of fracture and need utmost care while positioning during surgery. Traction or any corrective maneuver must be done carefully.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Braun J, Sieper J Ankylosing spondylitis. Lancet 2007;369:1379-90.  Back to cited text no. 1
    
2.
Belanger TA, Milam RA IV, Roh JS, Bohlman HH Cervicothoracic extension osteotomy for chin-on-chest deformity in ankylosing spondylitis. J Bone Joint Surg Am 2005;87:1732-8.  Back to cited text no. 2
    
3.
Ruf M, Rehm S, Poeckler-Schoeniger C, Merk HR, Harms J Iatrogenic fractures in ankylosing spondylitis—A report of two cases. Eur Spine J 2006;15:100-4.  Back to cited text no. 3
    
4.
Maruiwa R, Watanabe K, Suzuki S, Nori S, Tsuji O, Nagoshi N, et al. Chin on chest deformity caused by upper cervical kyphosis associated with ankylosing spondylitis: A case report. Neurospine 2020;17:666-71.  Back to cited text no. 4
    
5.
Wang H, Ding W-Y Ankylosing spondylitis with chin-on-chest deformity combined with Anderson lesion as the apex of cervicothoracic kyphosis: Case report. Int J Clin Exp Med2017;10:13831-5.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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