 |
 |
 |
|
| home | CME | help | feedback | subscriptions | archive |
|
© 1996 The Journal of Bone and Joint Surgery, Inc. Compression of the Spinal Cord Due to Destructive Spondyloarthropathy of the Atlanto-Axial Joints. A Case Report* , OKAYAMA, JAPANInvestigation performed at the Department of Orthopedic Surgery, Kawasaki Medical School, Okayama
Destructive spondyloarthropathy was apparently first described by Kuntz et al. in 1984 and has become increasingly recognized in patients who have been managed with long-term hemodialysis. Radiographically evident lesions are located predominantly in the cervical spine. The fifth and sixth and the sixth and seventh cervical intervertebral discs are most frequently involved13. Although destructive spondyloarthropathy affecting the occipitocervical articulation and the cephalad part of the cervical spine has been reported2,5,6,8,12,14, we are not aware of any previous reports of acute compression of the spinal cord attributed to these lesions. We report the case of a patient who had quadriplegia and dyspnea that were due to destructive spondyloarthropathy of the cephalad part of the cervical spine. Immediate reduction of the atlanto-axial dislocation and posterior arthrodesis of the atlanto-axial joint resulted in remarkable improvement.
A forty-five-year-old woman was admitted to our hospital in June 1994 because of progressive quadriplegia and dyspnea, which had begun twelve hours earlier. A nephrotic syndrome had developed when the patient was forty-two years old. The disease resulted in progressive deterioration of renal function requiring hemodialysis. Beginning in May 1991, the patient had received dialysis three times a week for three years. Pain and arthralgia became apparent for the first time in September 1993. The patient reported having pain in the shoulders, elbows, and thighs. In February 1994, pain developed in the posterior part of the neck and rapidly became severe. The diagnosis was destructive spondyloarthropathy at the level of the fifth and sixth cervical intervertebral disc. On June 30, 1994, the patient's husband raised the cephalad part of her trunk, while she was in bed, in order to alleviate the nuchal pain. The patient suddenly became quadriplegic with shooting pains from the nuchal region down both the upper and lower extremities. She was then admitted to a hospital. Radiographs of the cervical spine showed atlanto-axial dislocation with a fracture of the odontoid process (Fig. 1). Magnetic resonance images showed considerable compression of the spinal cord at the first and second cervical levels with slight compression of the cord at the fifth and sixth cervical levels (Fig. 2). The patient was then transferred to our hospital.
At the time of admission, the patient had complete quadriplegia. She reported pain in the nuchal region and difficulty with breathing. The trauma motor index16 was 1 of a possible 100 points, and the sensory level was at the fourth cervical vertebra. The biceps, triceps, patellar, and Achilles reflexes all were absent bilaterally with no plantar response. There was no sacral sparing. The arterial oxygen tension was sixty-eight millimeters of mercury (9.06 kilopascals) so nasal intubation was performed and the tension was maintained with intermittent positive-pressure ventilation. Application of skull traction with a halo device and three kilograms of weight resulted in good reduction of the atlanto-axial dislocation (Fig. 3). Three days later, the skull traction was replaced with a halo vest, and the trauma motor index16 improved to 49 points. Seven days later, the patient had recovered spontaneous breathing well enough for the nasal intubation to be withdrawn.
Radiographs revealed chondrocalcinosis of the elbows, wrists, hips, and knees. Analysis of crystal deposits aspirated from the elbow joint revealed that they were composed of hydroxyapatite. A computed tomography scan at the level of the odontoid process showed a calcification of the transverse atlantal ligament as well as destructive changes of the facet joints (Fig. 4). Laboratory evaluation of serum revealed a negative rheumatoid factor, a calcium level of 5.3 milliequivalents (2.65 millimoles) per liter (normal, 4.0 to 5.5 milliequivalents [2.00 to 2.75 millimoles] per liter), and a phosphorus level of 2.5 milliequivalents (1.4 millimoles) per liter (normal, 1.4 to 2.6 milliequivalents [0.8 to 1.4 millimoles] per liter). The level of alkaline phosphatase was 140 international units per liter (normal, twenty-five to eighty international units per liter), the beta-2-microglobulin was 41.6 micrograms per milliliter (3526 nanomoles per liter) (normal, 0.5 to 2.0 micrograms per milliliter [forty-two to 170 nanomoles per liter]), and the biologically active (intact) parathyroid hormone was 335 picograms per milliliter (35.3 picomoles per liter) (normal, 6.5 to 59.7 picograms per milliliter [0.7 to 6.3 picomoles per liter]). A technetium-99m bone scan showed increased uptake in the cranium and in the cephalad part of the cervical spine.
Posterior atlanto-axial arthrodesis was performed on July 21, 1994, with the patient wearing a halo vest (Fig. 5). Postoperatively, the patient recovered without incident and the neurological status improved gradually. On September 15, 1994, the patient had a trauma motor index16 of 96 points.
A disease known as dialysis arthropathy or destructive spondyloarthropathy can develop in patients who have severe chronic renal failure requiring long-term hemodialysis or peritoneal dialysis. Although erosive changes in the spine occur in as many as 25 per cent of patients being managed with dialysis (thirty of 118 in one series20), destructive changes are less common20. The frequency of dialysis arthropathy increases with the duration of the dialysis and approaches 90 per cent after ten years7. Hardouin et al. reported destructive spondyloarthropathy in 9 per cent of eighty patients who had had dialysis for a mean of 110 months. Kessler et al.13 reported the disorder in 5 per cent of forty patients who had had dialysis for more than ten years, and Iwamoto et al. reported it in 25 per cent of eighty-seven patients who had had dialysis for more than ten years. Other reports1,4 have indicated that dialysis is not required for the development of destructive spondyloarthropathy in patients who have chronic renal failure. Rather, the duration of renal failure may be more important4. Destructive spondyloarthropathy affects the cervical, lumbar, and thoracic spine in a ratio of approximately eighty-five to ten to five7. The caudad part of the cervical spine is the most frequent site of involvement. Abnormalities of the cervico-occipital articulation and the cephalad part of the cervical spine are unusual12. Although the pathophysiology of destructive spondyloarthropathy is not completely understood, several causative factors have been reported, including crystal-induced spondyloarthropathy11,15, amyloidosis associated with dialysis3,9, and secondary hyperparathyroidism1,17. Our patient may have sustained a pathological fracture of the odontoid process as a result of cystic lesions of bone from amyloidosis complicated by deposition of crystals in the transverse atlantal ligament and by secondary hyperparathyroidism. However, the exact etiology could not be determined, as no pathological studies were performed. The radiographic characteristics of destructive spondyloarthropathy are severe narrowing of the intervertebral space, erosions and geodes of adjacent vertebral plates, and the absence of notable osteophytosis. It is usually difficult to differentiate destructive spondyloarthropathy from infection with use of plain radiographs alone. Modic et al. reported that, on magnetic resonance imaging, the findings of vertebral osteomyelitis consisted of a decreased signal intensity from the vertebral bodies and the intervening discs on T1-weighted images. On T2-weighted images, there was an irregularly increased signal intensity from the disc and the adjacent end plates. Rafto et al. found low signal intensity on both T1 and T2-weighted images of patients who had destructive spondyloarthropathy. They stated that an inflammatory response causes signal characteristics on a magnetic resonance image that may be indistinguishable from those of an infection. Diagnosis of destructive spondyloarthropathy may not be difficult if the frequency of the disorder among patients who have chronic renal failure is appreciated. Evidence of amyloid-related disease is often found on the radiographs of asymptomatic patients who are being managed with dialysis. Treatment is needed when there are signs or symptoms of compression of a nerve root or of the spinal cord. The unpredictable but often rapid nature of the disease makes operative decompression and stabilization necessary in some patients7. An internist, nephrologist, pathologist, or radiologist is often the first physician to confront this disease clinically. However, orthopaedic surgeons also should consider destructive spondyloarthropathy in the differential diagnoses of spinal conditions in patients who have chronic renal failure.
*No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. No funds were received in support of this study.
  CiteULike  Connotea  Del.icio.us  Facebook  Technorati  Twitter What's this?
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Top
Introduction
Case Report
| home | CME | help | feedback | subscriptions | archive | site index |
|
Stanford University Libraries' HighWire Press (TM) assists in the publication of JBJS Online. |