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Year : 2020  |  Volume : 3  |  Issue : 3  |  Page : 97-100

Heterotopic ossification in a patient with cervical spinal cord injury and ankylosing spondylitis: The consequences of the late diagnosis

Spinal Injury Unit, Royal National Orthopaedic Hospital, Stanmore, England, UK

Date of Submission04-May-2020
Date of Decision06-Jun-2020
Date of Acceptance15-Jun-2020
Date of Web Publication24-Jul-2020

Correspondence Address:
Dr. Celine Lakra
Royal National Orthopaedic Hospital, Brockley Hill, Stanmore HA7 4LP
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jisprm.jisprm_20_20

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Patients with ankylosing spondylitis (AS) are at an 11-fold greater risk of vertebral fracture, with higher mortality and morbidity rates than the general spinal cord injury (SCI) population. We present the case of a patient with established AS who suffered from a cervical SCI. Following admission to a specialist spinal injury rehabilitation unit, he was diagnosed with severe established heterotopic ossification (HO) and spasticity. The key considerations required to manage a case of significant disability in the rehabilitation setting are outlined. The need for earlier diagnosis of HO to prevent progression and associated complications is discussed.

Keywords: Ankylosing spondylitis, disability, heterotopic ossification, spinal cord injury

How to cite this article:
Lakra C, Desai M. Heterotopic ossification in a patient with cervical spinal cord injury and ankylosing spondylitis: The consequences of the late diagnosis. J Int Soc Phys Rehabil Med 2020;3:97-100

How to cite this URL:
Lakra C, Desai M. Heterotopic ossification in a patient with cervical spinal cord injury and ankylosing spondylitis: The consequences of the late diagnosis. J Int Soc Phys Rehabil Med [serial online] 2020 [cited 2023 May 28];3:97-100. Available from: https://www.jisprm.org/text.asp?2020/3/3/97/290576

  Introduction Top

Patients with ankylosing spondylitis (AS) are at an 11-fold greater risk of vertebral fracture,[1] commonly caused by falls, with 81% occurring in the cervical spine.[2] Overall mortality is high, at 35%–50% in patients with spinal cord injury (SCI) and AS, compared to 4%–16% in patients with without AS.[1] The risk of secondary neurological deficit following vertebral fracture is also considerably higher.[3]

Only one study has analyzed the relationship between AS and developing heterotopic ossification (HO) following SCI.[4] This concludes that patients with AS are not at increased risk (relative risk = 1.16; 95% confidence interval = 0.65–2.09), but the risk almost doubles in the presence of complete spinal injury or pressure ulceration.

Other studies conclude that the development of HO following SCI is associated with preventable outcomes, such as spasticity, pneumonia, presence of a tracheostomy, and urinary tract infection.[5],[6],[7] Whether such outcomes are risk factors or alternatively complications of HO has yet to be studied.

There is no published literature demonstrating a relationship between the development of HO following SCI in patients with leukemia, diabetes, or hypertension.

  Case Report Top

A 54-year-old male was admitted following a fall backward. He developed acute loss of sensation and weakness in the upper and lower limbs. He had a background of AS, chronic myeloid leukemia, diabetes, and hypertension.

A computed tomography (CT) of the neck [Figure 1] demonstrated the classical features of AS, including calcification of the vertebral bodies and severe ossification of the anterior and posterior longitudinal ligaments. Tiny bone fragments around C1, the dens of C2, and a C2 osteophyte fracture were highlighted. Further imaging [Figure 2] demonstrated significant multilevel spinal canal stenosis with compression of the cord, most severe at the level of C2/3. In the acute hospital, the patient was managed conservatively with a collar for 6 weeks.
Figure 1: Emergency computed tomography of the neck demonstrated the classical features of ankylosing spondylitis. Tiny fragments around C1 and the dens of C2 and a probable C2 osteophyte fracture were highlighted

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Figure 2: Magnetic resonance imaging of the neck demonstrating multilevel spinal canal stenosis with compression of the cord, most severe at C2/3

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After a 3-month long admission on the acute spinal ward, he was transferred to a highly specialized multidisciplinary rehabilitation unit for spinal cord-injured patients. At this stage, he was classified as having a C4B injury (International Standards for the Neurological Classification of SCI[8]). On admission, he was noted to have severe spasticity and spasms in his lower limbs. Further assessment revealed a locked position of the hip joints in all movement planes fixed at 40° flexion, associated with pain. X-ray of the hips and pelvis confirmed the diagnosis of severe bilateral HO [Figure 3]. This had not been recognized during his acute admission.
Figure 3: X-ray demonstrating severe bilateral heterotopic ossification of the hips following spinal cord injury

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Given the significant restriction of the hip movement and associated spasticity, ongoing management in the rehabilitation setting required a considered and coordinated multidisciplinary approach. Medical management included optimizing pain, spasms, and spasticity. Baclofen, tizanidine, amitriptyline, gabapentin, and indomethacin were commenced. The patient suffered with severe drowsiness that adversely affected his ability to participate actively in rehabilitation. Medications were, therefore, carefully tapered to provide an effective treatment dose while limiting such side effects.

The patient went on to have bilateral CT-guided steroid injections of the hips. CT demonstrated the extent of disease [Figure 4]. While the procedure significantly improved his pain, the patient's hip range of motion and position remained unchanged.
Figure 4: Computed tomography confirms the diagnosis of severe heterotopic ossification affecting the hip joints

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A bone scan 5 months after the initial spinal injury revealed intensely active HO in both of his hips and iliac bones [Figure 5]. Indomethacin was therefore thought to have been ineffective at treating HO and hence was stopped. He was further treated with a zoledronic acid infusion, in line with the current evidence.[9]
Figure 5: Bone scan revealed intensely active heterotopic ossification in the hips and iliac bones

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Therapy goals focused on maintaining and increasing the limited range of movement of the hips to improve positioning in bed and considering options available for sitting upright. This was achieved through passive stretching, postural management, and the development of a personalized sleep system [Figure 6].
Figure 6: A personalized sleep system was initiated to maintain the patient's position when supine

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A specially configured tilt-in-space wheelchair with a 50° recline and a high-pressure relieving cushion was manufactured and provided on discharge [Figure 7]. Delivering appropriate seating and sleep systems were extremely challenging given the patient's posture; however, with this management, his skin integrity was maintained. On discharge, despite vigorous input from the multidisciplinary team, the patient's neurological status and posture remained similar to that of admission.
Figure 7: A specially configured tilt-in-space wheelchair with a 50° recline and a high-pressure relieving cushion was provided. He has resting wrist splints in place, as well as a cough-assist device which also acts as his call bell. Positioning guidelines and a hoist are also in view

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  Discussion Top

On admission to the unit, this patient was found to have developed serious and limiting complications as a direct result of his SCI occurring 3 months prior. These had not been recognized by the medical, surgical, or therapy teams in the acute setting. Once diagnosed, the patient's resulting disability was already significant and largely irreversible. Rehabilitation therefore focused on preventing further decline and managing significant disability.

Early recognition of HO in the acute setting could result in better outcomes, reduced morbidity, and reduced care costs. Prevention and early detection of risk factors and complications could reduce the incidence and progression of HO, respectively. Further longitudinal research is required to confirm if associated outcomes such as spasticity cause or complicate HO.

The authors recommend that acute trauma and spinal units include specialists in rehabilitation medicine as an integral component of the clinical team to manage such conditions early on in their course. This is in line with the national recommendations[10] however is seldom implemented.

Declaration of patient consent

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

Financial support and sponsorship


Conflicts of interest

There are no conflicts of interest.

  References Top

Jacobs WB, Fehlings MG. Ankylosing spondylitis and spinal cord injury: Origin, incidence, management, and avoidance. Neurosurg Focus 2008;24:E12.  Back to cited text no. 1
Westerveld LA, Verlaan JJ, Oner FC. Spinal fractures in patients with ankylosing spinal disorders: A systematic review of the literature on treatment, neurological status and complications. Eur Spine J 2009;18:145-56.  Back to cited text no. 2
Ahmad K, Matee S, Bin Ayaz S, Rashid Ihsan H. Spinal cord injury-an unusual presentation for advanced asymptomatic ankylosing spondylitis. Acta Neurol Taiwan 2015;24:102-5.  Back to cited text no. 3
Ohlmeier M, Karras P, Suero EM, Grasmücke D, Aach M, Meindl R, et al. Ankylosing spondylitis does not increase the risk of neurogenic heterotopic ossification in patients with a spinal cord injury: A retrospective cohort study. Spinal Cord 2017;55:213-5.  Back to cited text no. 4
Citak M, Suero EM, Backhaus M, Aach M, Godry H, Meindl R, et al. Risk factors for heterotopic ossification in patients with spinal cord injury: A case-control study of 264 patients. Spine (Phila Pa 1976) 2012;37:1953-7.  Back to cited text no. 5
Coelho CV, Beraldo PS. Risk factors of heterotopic ossification in traumatic spinal cord injury. Arq Neuropsiquiatr 2009;67:382-7.  Back to cited text no. 6
Sullivan MP, Torres SJ, Mehta S, Ahn J. Heterotopic ossification after central nervous system trauma: A current review. Bone Joint Res 2013;2:51-7.  Back to cited text no. 7
Kirshblum SC, Burns SP, Biering-Sorensen F, Donovan W, Graves DE, Jha A, et al. International standards for neurological classification of spinal cord injury (revised 2011). J Spinal Cord Med 2011;34:535-46.  Back to cited text no. 8
Teasell RW, Mehta S, Aubut JL, Ashe MC, Sequeira K, Macaluso S, et al. A systematic review of the therapeutic interventions for heterotopic ossification after spinal cord injury. Spinal Cord 2010;48:512-21.  Back to cited text no. 9
British Society of Rehabilitation Medicine, Specialist Rehabilitation in the Trauma Pathway. BSRM Core Standards, United Kingdom; 2018.  Back to cited text no. 10


  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


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