Cervical DISH, is there any place for multilevel cervical arthroplasty? A case report metal-on-metal two-level cervical disc arthroplasty with 2-year follow-up and quality of life assessment

Abstract

Introduction and importance:

Cervical diffuse idiopathic skeletal hyperostosis (DISH) can cause stiffness, reducing quality of life as the result of functional impairment and progressive pain. The standard treatment depends on patient’s problem which often involves anterior cervical discectomy and function, but this approach can lead to ASD. Cervical disc arthroplasty (CDA) using a metal-on-metal (MoM) prosthesis may offer an alternative treatment to preserve segmental motion while alleviating symptoms.

Case presentation:

A 55-year-old man presented with progressive cervical stiffness and pain, which reduced his quality of life and worsened in the past 2 years. The lateral cervical radiograph revealed ossification of the anterior longitudinal ligament in the anterolateral area from C2 until C7. The CT scan showed ossification from C2 to T6. Attempted conservative treatment had failed. The patient underwent two-level CDA MoM at C4-5 and C5-6.

Clinical discussion:

The patient experienced significant improvements in pain (VAS reduced from 5/10 to 0/10). At the 12- and 24- month postoperative follow-ups, there is an improvement in the Neck Disability Index (NDI) and SF-36 score. Clinical function (by NDI) and the physical and mental components of SF-36 were parameters with significant improvement, which led to patient satisfaction. No implant-related complications or ASD were observed at the 2-year follow-up.

Conclusion:

This case highlights the potential benefits of MoM two-level CDA for selected DISH patients with degenerative disc disease. The procedure preserved mobility, reduced pain, and improved quality of life without ASD. Further studies are needed to evaluate long-term outcomes.

Introduction

Diffuse idiopathic skeletal hyperostosis (DISH) is a non-inflammatory spinal disorder characterized by continues ossification of the anterior longitudinal ligament (OALL), leading to stiffness and pain. Although commonly asymptomatic, DISH involving the cervical spine can cause significant neck pain, reduced range of motion (ROM), dysphagia, and neurological symptoms due to compression of adjacent structures, resulting in reduced quality of life.

HIGHLIGHTS

• DISH is a systemic disorder with spinal ligament ossification.

• Cervical stiffness and pain reduce patient quality of life.

• Two-level cervical disc arthroplasty preserved motion and improved outcomes.

The standard surgical treatment for symptomatic DISH with degenerative disc disease is anterior cervical discectomy and fusion. However, fusion procedures are associated with increased stress on adjacent segments, leading to adjacent segment disease (ASD) over time. Cervical disc arthroplasty (CDA) is an alternative that preserves motion, reduces biomechanical stress on adjacent segments, and maintains cervical alignment.

This case report presents a 55-year-old man with cervical DISH and degenerative disc disease at C4-5 and C5-6, successfully treated with metal-on-metal (MoM) two-level cervical disc replacement. Clinical outcomes, pain levels, and quality of life were evaluated over a 2-year follow-up period. Our work has been reported in line with the Surgical Case Report (SCARE) Guidelines[1].

Presentation of case

A 55-year-old man was admitted with progressive neck stiffness and pain over 2 years. He cannot turn aside and rotate his neck. He was significantly hindered in daily activities such as turning to the side when somebody called or driving a vehicle. There were no symptoms such as radiculopathy, dysphagia, neurological deficits, and gait disturbances. There was no history of inflammatory arthritis, trauma, or previous spinal surgery.

A physical examination on admission demonstrated an almost totally decreased neck ROM. Flexion/extension was 10/5°, with no rotation or lateral flexion. The neck pain was VAS 5/10, and there was no arm pain. There was no neurologic deficit or gait disturbance present. The Neck Disability Index (NDI) preoperative was 36%, with the biggest components being driving (4), recreation (4), lifting (3), reading (2), personal care (2), working (2), and pain (1). SF-36 was done preoperatively with the results: physical functioning 40%, energy 60%, emotional well-being 68%, social functioning 50%, pain 45%, general health 65%, and health change 50%.

The lateral cervical radiograph showed an extensive OALL from C2 until thoracic 6 with a thickness of 1.2–1.8 mm (Fig. 1). A computed tomography (CT) scan showed ossification in anterior from C2 until mid-thoracic (Th 6) and spinal canal narrowing and no OPLL (Fig. 2). The magnetic resonance imaging (MRI) showed disc degeneration with reduced relative normal disc height, normal facets, no spinal canal narrowing, and no cord compression (Fig. 3).

Figure 1.

Cervical X-ray in lateral position shows ossification of ALL from Tip C2 until lower cervical, with 1.8 mm thickness.

Figure 2.

A 3D CT scan non-contrast image. A thick flowing ossification of the anterior lateral ligament is shown at the sagittal image. (A). The coronal view showed the extend of ossification (B).

Figure 3.

A non-contrast cervical MRI showed (A) no canal stenosis, DDD, and (B) normal facet joint in axial view.

Because the facet and disc were still good as proven by preoperative radiographs as well as intra-operative evaluation, the CDA procedure was conducted under intraoperative neuromonitoring. Two levels CDA at C4-5 and C5-6 (two levels considerations due to financial limitation) were done with MoM type of disc replacement using Activ C intervertebral disc prosthesis (B.Braun, Sheffield, UK) (Fig. 4).

Figure 4.

(A) Postoperative X-ray at the latest follow-up showed that the facets condition is still relatively good with minimal degeneration. (B) Postoperative neck rotations are preserved.

The patient was discharged after 1 day of postoperative observation. No complication was noticed at the perioperative period and at the postoperative follow-up. After surgery, the patient regained his neck ROM: Flexion/extension 35/25°, right/left rotation 60/45°, and right/left lateral flexion 30/25° (Table 1). The NDI was improving at the 3-month follow-up and stable up to the last follow-up (Table 2). The components of NDI that changed after surgery are lifting (1), driving (2), reading (1), and recreation (2). The SF-36 showed significant quality of life improvement (Table 3), and the patient rates 5 for the patient satisfaction scale (very satisfied).

Table 1.

Neck range of motion pre- and post-surgery.

		Postoperative
	Pre-op	3 months	6 months	1 year	2 years
Flexion/extension	10/5	35/25	35/25	35/25	35/25
R/L rotation	0	60/45	60/45	60/45	60/45
R/L lateral F	0	30/25	30/25	30/25	30/25

Table 2.

Patient quality of life measurement pre- and post-surgery.

Timepoint	VAS (pain score)	NDI (disability index)	Neck ROM	SF-36 QoL score
Pre-op	5/10	36%	Limited	Low
3 months	1/10	12%	Improved	Improved
6 months	0/10	12%	Improved	Improved
12 months	0/10	12%	improved	Significantly Improved
24 months	0/10	12%	Improved	Stable Improvement

Table 3.

SF-36 score pre- and post-surgery.

SF-36 components	Pre-op	After 2 years
Physical functioning	40%	90%
Energy	60%	100%
Social functioning	50%	75%
pain	45%	87.5%
General health	65%	55%
Health changes	50%	100%

Discussion

DISH was originally described in 1950 by Forestier and Rotes-Querol. The most widely accepted radiologic criteria for DISH were proposed by Resnick and Niwayama^[2–4^] in 1976 which include the following: Calcification along the anterolateral aspect at least four contiguous vertebral bodies, relative preservation of intervertebral disc height in affected areas, and absence of apophyseal joint bone ankylosis, and sacroiliac joint erosion, sclerosis, or intraarticular osseous fusion. In this case, the patient met the criteria with the ossification involved more than four levels, and the disc condition was still good, with no facet degeneration, and the diagnosis of DISH was built^[5,6^].

DISH occurs frequently in middle-aged and elderly man, which suited to our patient characteristic. In general population, the prevalence is estimated between 6% and 12% on the basis of autopsy series and is often related to OPLL in 50% of cases. This fact also applies to our patient, who does not have OPLL.

The majority of DISH are asymptomatic^[7–9^]. The most common presenting symptoms of cervical DISH are dysphagia. Dyspnea and neurologic symptoms are less frequent, occurring in only 4% of cases. In this case, the presenting symptoms are neck stiffness and pain, without neurological symptoms. The neurological deficit affecting 4% of patients with DISH, which more likely caused by cervical stenosis, atlanto-axial subluxation, or trivial trauma on OPLL^[10–12^].

The lateral X-ray showed ossification along anterolateral margin from C2 until mid-thoracic with variable thickness which met the criteria for cervical DISH. The MRI was done to assess the condition of the disc, end plate, spinal canal, and posterior area of the cervical including facet, and we found that the condition of disc and facet was good. CT scan confirmed the condition of facet and the extend of ossification^[13,14^].

The extensive OALL in this patient is indeed raising an important consideration, as such a pattern typically shows ankylosing tendencies and is commonly associated with reduced segmental mobility which leads to a progressive spinal stiffness. Conventionally, that condition is considered as a relative contraindication to motion-preserving procedures due to the concerns about spontaneous fusion, heterotopic ossificans, and loss of long-term prosthesis mobility. However, the evidence of preservation of segmental motion as proven with the good facet and disc (the mobile segment), which is further proven by intraoperative condition are the main reason to choose arthroplasty to improve some motion of the important area which increasing part of motion in rotation flexion extension and lateral bending, not all the segments from C2 until C7, and thereby increase quality of life. Nevertheless, given the progressive nature of OALL, long-term maintenance of motion cannot be assumed, and careful patient selection and continued radiographic surveillance remain essential[15].

The CDA level in this patient were C4-5 and C5-6 segments base on the consideration that those levels are second most important level (after C1-C2) for flexion-extension, lateral bending, and also contributing in axial rotation[16]. The preservation of motion which is the benefit of this surgery has proved that the ROM improvement significantly affect the other quality of life scores along with the pain level improvement. These achievements are also reported by Obernaueur et al[17] who used the CDA using MoM in degenerative cases.

The neck pain was significantly improved from 5 to 1 after 3 months post-surgery and become painless (0) after 6 months until the latest follow-up. This result is also the same as other literature that using MoM CDA in other cases[17]. SF-36 showed that after 2 years, physical functioning increased significantly, energy and health changes got better, and also quality of life improved as social functioning got better.

The prosthesis used in this patient is MoM-type CDA. Although several MoM designs have been withdrawn in certain regions, we still use that kind of CDA in Indonesia. In this patient, we used Active-C® that consists of a superior cobalt–chromium–molybdenum endplate with anchoring spikes and inferior endplate with the same material incorporating a central anchoring fin and an integrated ultra-high-molecular-weight polyethylene inlay. With respect to concerns regarding the metal wear, it is important to highlight that the implant design is not a pure MoM articulation, as motion occurs between cobalt–chromium endplates and a polyethylene inlay. Thus, reducing the risk of metal ion release leads to metal wear as seen in earlier MoM design. Clinical studies by McGonagle et al[18] reported a favorable result in series of cervical myelopathy treated with this kind of MoM implant. However, given the issue about the metal wear, it is wise to continuously observe the patient’s clinical and radiograph in a long-term follow-up.

There was no new ossification in anterolateral of C4-5, C5-6 after 2 years of surgery, and there was no implant dislodge, clicking, or other symptoms at the latest follow-up.

Conclusion

This case showed that that MoM two-level cervical disc replacement is a viable option for select DISH patients, with right choices level will increase quality of life. The procedure maintained segmental mobility, reduced pain, and improved quality of life over 2 years, with no adjacent segment degeneration. Further studies should focus on a long-term outcome, implant wear, and potential complications in patients undergoing CDA for cervical DISH with more cases.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Consent

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Efforts have been made to ensure anonymity; the authors confirm that the patient’s identity has been protected.

Sources of funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Conflicts of interest disclosure

None declared.

Author contributions

All authors contributed to data analysis, drafting or revising the article, have agreed on the journal to which the article will be submitted, gave final approval of the version to be published, and agree to be accountable for all aspects of the work.

Guarantor

Didik Librianto.

Research registration unique identifying number (UIN)

Not applicable.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Data availability statement

The data that support the findings of this study are available from the corresponding author upon reasonable request.

Disclaimer

No author details are included in the figures.