JBJS | Compression of the Spinal Cord Due to Destructive Spondyloarthropathy of the Atlanto-Axial Joints. A Case Report*

The Journal of Bone & Joint Surgery, Volume 78, Issue 12

Articles | December 01, 1996

Compression of the Spinal Cord Due to Destructive Spondyloarthropathy of the Atlanto-Axial Joints. A Case Report*

YOSHIHIRO MIKAWA, M.D.†; TOSHIO YAMAOKA, M.D.†; RYO WATANABE, M.D.†, OKAYAMA, JAPAN

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Investigation performed at the Department of Orthopedic Surgery, Kawasaki Medical School, Okayama

The Journal of Bone & Joint Surgery. 1996; 78:1911-4

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Introduction

Introduction | Case Report | Discussion | References

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 reported^{2,5,6,8,12,14}, 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.

*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.

†Department of Orthopedic Surgery Kawasaki Medical School, 577 Matsushima, Kurashiki, Okayama 701-01, Japan.

*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.

†Department of Orthopedic Surgery Kawasaki Medical School, 577 Matsushima, Kurashiki, Okayama 701-01, Japan.

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Fig. 1 Lateral radiograph of the cervical spine, made at the referring hospital, showing atlanto-axial dislocation with a fracture of the odontoid process.

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Fig. 2 Sagittal T2-weighted magnetic resonance image of the cervical spine, made at the referring hospital, showing marked compression of the spinal cord at the first and second cervical levels with concomitant destructive spondyloarthropathy at the fifth and sixth cervical levels.

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Fig. 3 Lateral tomogram of the cephalad part of the cervical spine, showing good reduction of the atlanto-axial dislocation and the pathological fracture of the odontoid process.

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Fig. 4 Computed tomography scan through the atlas, showing destructive changes of the first cervical body and the odontoid process with deposition of crystals in the transverse atlantal ligament.

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Fig. 5 Intraoperative radiograph showing the site of the posterior atlanto-axial arthrodesis.

Case Report

Introduction | Case Report | Discussion | References

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.

Discussion

Introduction | Case Report | Discussion | References

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 reports^1,4 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 spondyloarthropathy^11,15, amyloidosis associated with dialysis^3,9, and secondary hyperparathyroidism^1,17. 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.

References

Introduction | Case Report | Discussion | References

Alcalay, M.; Goupy, M. C.; Azais, I.; and |and |Bontoux, D.: Hemodialysis is not essential for the development of destructive spondylarthropathy in patients with chronic renal failure. Arthrit. and Rheumat.,30, 182-1186: 1987.182-118630 1987

Allard, J. C.; Artze, M. E.; Porter, G.; Ghandur-Mnaymneh, L.; de Velasco, R.; and |and |Perez, G. O.: Fatal destructive cervical spondyloarthropathy in two patients on long-term dialysis. Am. J. Kidney Dis.,19: 81-85, 1992.1981 1992 [PubMed]

Bardin, T.; Zingraff, J.; Shirahama, T.; Noel, L. H.; Droz, D.; Voisin, M. C.; Drueke, T.; Dryll, A.; Skinner, M.; Cohen, A. S.; and |and |Kuntz, D.: Hemodialysis-associated amyloidosis and beta-2 microglobulin. Clinical and immunohistochemical study. Am. J. Med.,83: 419-424, 1987.83419 1987 [PubMed][CrossRef]

Bindi, P., and |and |Chanard, J.: Destructive spondyloarthropathy in dialysis patients: an overview. Nephron,55: 104-109, 1990.55104 1990 [PubMed][CrossRef]

Chassagne, P.; Said, L. A.; Le Loet, X.; Deshayes, P.; Noel, L. H.; Dhib, M.; Joannides, R.; Geffroy-Josse, S.; and |and |Godin, M.: Fatal destructive cervical spondyloarthropathy [letter]. Am. J. Kidney Dis.,20: 99-200, 1992.2099 1992

Corinaldesi, A.; Zompatori, M.; Pisi, P.; Ambrosetto, P.; Canini, R.; Mosconi, G.; Rimondi, M. R.; and |and |Orlandi, P. E.: La spondilopatia destruente del dializzato. Ruolo diagnostico della Risonanza Magnetica. Radiol. Med. (Torino),84: 221-227, 1992.84221 1992

Davidson, G. S.; Montanera, W. J.; Fleming, J. F.; and |and |Gentili, F.: Amyloid destructive spondyloarthropathy causing cord compression: related to chronic renal failure and dialysis. Neurosurgery,33: 519-522, 1993.33519 1993 [PubMed][CrossRef]

Gerster, J. C.; Carruzzo, P. A.; Ginalski, J. M.; and |and |Wauters, J. P.: Cervicooccipital hinge changes during longterm hemodialysis. J. Rheumatol.,16: 1469-1473, 1989.161469 1989 [PubMed]

Hardouin, P.; Flipo, R. M.; Foissac-Gegoux, P.; Thevenon, A.; Pouyol, F.; Duquesnoy, B.; and |and |Delcambre, B.: Current aspects of osteoarticular pathology in patients undergoing hemodialysis: study of 80 patients. Part 1. Clinical and radiological analysis. J. Rheumatol.,14: 780-783, 1987.14780 1987 [PubMed]

Iwamoto, N.; Tanaka, T.; Kawase, Y.; Yamazaki, S.; Kondo, M.; Hiratake, Y.; Miyamoto, T.; Imai, R.; Yamamoto, N.; Fukuda, T.; and |and |Ono, T.: Clinical study of destructive spondylarthropathy of the cervical spine in patients on chronic (10-year or longer) hemodialysis. J. Japanese Soc. Dial. Ther.,23:: 299-305, 1990.23:299 1990

Kaplan, P.; Resnick, D.; Murphey, M.; Heck, L.; Phalen, J.; Egan, D.; and |and |Rutsky, E.: Destructive noninfectious spondyloarthropathy in hemodialysis patients: a report of four cases. Radiology,162: 241-244, 1987.162241 1987 [PubMed]

Kessler, M.; Netter, P.; Grignon, B.; Bertheau, J. M.; Aymard, B.; Azoulay, E.; and |and |Gaucher, A.: Destructive beta 2-microglobulin amyloid arthropathy of the cervico-occipital hinge in a hemodialyzed patient [letter]. Arthrit. and Rheumat.,33: 602-604, 1990.33602 1990 [CrossRef]

Kessler, M.; Netter, P.; Delons, S.; Grignon, B.; Cao Huu, T.; Chanliau, J.; Pere, P.; Poignet, J. L.; Benoit, J.; and |and |Gaucher, A.: Complications articulaires chez les insuffisants rénaux chroniques hémodialysés depuis plus de 10 ans. Quarante cas. Presse méd.,17: 679-682, 1988.17679 1988 [PubMed]

Kröoner, G.; Stäler, A.; Seiderer, M.; Moran, J. E.; and |and |Gurland, H. J.: Beta 2 microglobulin-related amyloidosis causing atlantoaxial spondylarthropathy with spinal-cord compression in aae patients: detection by MRI. Nephrol., Dial., Transplant.,6 (Supplement 2): 91-95, 1991.6 (Supplement 2)91 1991

Kuntz, D.; Nevi, B.; Bardin, T.; Drueke, T.; Treves, R.; and |and |Dryll, A.: Destructive spondylarthropathy in hemodialyzed patients. A new syndrome. Arthrit. and Rheumat.,27: 369-375, 1984.27369 1984 [CrossRef]

Lucas, J. T., and |and |Ducked, T. B.: Motor classification of spinal cord injuries with mobility, morbidity and recovery indices. Am. Surg.,45: 151-158, 1979.45151 1979 [PubMed]

McCarthy, J. T.; Dahlberg, P. J.; Kriegshauser, J. S.; Valente, R. M.; Swee, R. G.; O'Duffy, J. D.; Kurtz, S. B.; and |and |Johnson, W. J.: Erosive spondyloarthropathy in long-term dialysis patients: relationship to severe hyperparathyroidism. Mayo Clin. Proc.,63: 446-452, 1988.63446 1988 [PubMed]

Modic, M. T.; Feiglin, D. H.; Piraino, D. W.; Boumphrey, F.; Weinstein, M. A.; Duchesneau, P. M.; and |and |Rehm, S.: Vertebral osteomyelitis: assessment using MR. Radiology,157: 157-166, 1985.157157 1985 [PubMed]

Rafto, S. E.; Dalinka, M. K.; Schiebler, M. L.; Burk, D. L., Jr.; and |and |Kricun, M. E.: Spondyloarthropathy of the cervical spine in long-term hemodialysis. Radiology,166: 201-204, 1988.166201 1988 [PubMed]

Sundaram, M.; Seelig, R.; and |and |Pohl, D.: Vertebral erosions in patients undergoing maintenance hemodialysis for chronic renal failure. AJR. Am. J. Roentgenol.,149: 323-327, 1987.149323 1987 [PubMed]

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Fig. 1 Lateral radiograph of the cervical spine, made at the referring hospital, showing atlanto-axial dislocation with a fracture of the odontoid process.

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Fig. 3 Lateral tomogram of the cephalad part of the cervical spine, showing good reduction of the atlanto-axial dislocation and the pathological fracture of the odontoid process.

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Fig. 4 Computed tomography scan through the atlas, showing destructive changes of the first cervical body and the odontoid process with deposition of crystals in the transverse atlantal ligament.

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Fig. 5 Intraoperative radiograph showing the site of the posterior atlanto-axial arthrodesis.

Bindi, P., and |and |Chanard, J.: Destructive spondyloarthropathy in dialysis patients: an overview. Nephron,55: 104-109, 1990.55104 1990 [PubMed][CrossRef]

Gerster, J. C.; Carruzzo, P. A.; Ginalski, J. M.; and |and |Wauters, J. P.: Cervicooccipital hinge changes during longterm hemodialysis. J. Rheumatol.,16: 1469-1473, 1989.161469 1989 [PubMed]

Lucas, J. T., and |and |Ducked, T. B.: Motor classification of spinal cord injuries with mobility, morbidity and recovery indices. Am. Surg.,45: 151-158, 1979.45151 1979 [PubMed]

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YOSHIHIRO MIKAWA, TOSHIO YAMAOKA, RYO WATANABE; Compression of the Spinal Cord Due to Destructive Spondyloarthropathy of the Atlanto-Axial Joints. A Case Report*. The Journal of Bone & Joint Surgery. 1996 Dec;78(12):1911-4.

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