Abstract
Ankylosing spondylitis (AS) is an autoimmune spondyloarthropathy involving principally the sacroiliac joint and axial skeleton. Spinal cord involvement is an infrequent and late complication. It mostly results from compressive myelopathy due to skeletal osteopathy and usually presents with radiculomyelopathic sensory and motor deficits. To report three patients who suffered a progressive paraparesis/tetraparesis compatible with motor myelopathy without typical skeletal symptom. Myelopathy of unknown origin was initially interpreted in these patients. Radiography did not show typical change at sacroiliac joint or vertebrate. Spinal magnetic resonance image revealed cord atrophy at cervical and thoracic segment. A positivity of B27 antigen was found afterward. Their spondyloarthropathic symptoms developed within six months later with radiographic sacroiliitis. Seropositive AS with noncompressive myelopathy was finally established. Patients showed a reverse of motor impairment when their pain was well undercontrolled. Motor myelopathy may be neglected or underestimated in AS, in especially when typical skeletal symptom is absent or minimal. It may progress surreptitiously to harm spinal function or superimpose to crippling disability in compressive spinal cord injury. Therefore, a careful evaluation and monitor of spinal cord function is important for AS patient despite spinal deformity is not observed.
References
Braun J, Sieper J (2007) Ankylosing spondylitis. Lancet 369(9570):1379–1390
Hunter T (1989) The spinal complications of ankylosing spondylitis. Semin Arthritis Rheum 19:172–182
Matthews WB (1968) The neurological complications of ankylosing spondylitis. J Neurol Sci 6:561–573
Rosenbaum RB (2000) Disorders of bones joints, ligaments and meninges. In: Bradley WG, Daroff RB, Fenichel GM, Marsden CD (eds) Neurology in Clinical Practise, The Neurological Disorders, vol 77, 3rd edn. Butterworth-Heinemann, USA, pp 1953–1984
Westerveld LA, Verlaan JJ, Oner FC (2009) Spinal fractures in patients with ankylosing spinal disorders: a systematic review of the literature on treatment, neurological status and complications. Eur Spine J 18:145–156
Jacobs WB, Fehlings MG (2008) Ankylosing spondylitis and spinal cord injury: origin, incidence, management, and avoidance. Neurosurg Focus 24:E12
Nordqvist L (1964) The sagittal diameter of the spinal cord and subarachnoid space in different age groups. A roentgenographic post-mortem study. Acta Radiol 227(Suppl):1–96
Tan FU, Tellioğlu S, Aydin G, Erdemoğlu AK, Keleş I (2004) Ankylosing spondylitis and multiple sclerosis in an HLA-B27 negative patient. Acta Neurol Belg 104:169–172
Khan MA, Kushner I (1979) Ankylosing spondylitis and multiple sclerosis. Arthritis Rheum 22:784–786
Hanrahan PS, Russell AS, McLean DR (1988) Ankylosing spondylitis and multiple sclerosis: an apparent association? J Rheumatol 15:1512–1514
Whitman GJ, Khan MA (1989) Unusual occurrence of ankylosing spondylitis and multiple sclerosis in a black patient. Cleve Clin J Med 56:819–822
Libbrecht N, De Bleecker J (1999) Ankylosing spondylitis and multiple sclerosis. Acta Clin Belg 54:30–32
Cellerini M, Gabbrielli S, Bongi SM (2001) Cerebral magnetic resonance imaging in a patient with ankylosing spondylitis and multiple sclerosis syndrome. Neuroradiology 43:1067–1069
Zwecker M, Daich A, Blumen N, Zeilig G, Ohry A (2000) Slow ascending myelopathy, tetraplegia, carcinoma of the bladder and amyloidosis in a patient with ankylosing spondylitis. Spinal Cord 38:327–329
Foo D, Bignami A, Rossier AB (1983) Two spinal cord lesions in a patient with ankylosing spondylitis and cervical spine injury. Neurology 33:245–249
Thomas DJ, Kendall MJ, Whitfield AGW (1974) Nervous system involvement in ankylosing spondylitis. BMJ 1:148–150
Dolan AL, Gibson T (1994) Intrinsic spinal cord lesions in 2 patients with ankylosing spondylitis. J Rheumatol 21:1160–1161
Pillay N, Hunter T (1976) Delayed evoked potentials in patients with ankylosing spondylitis. J Rheumatol 13:137–141
Khedr EM, Rashad SM, Hamed SA, El-Zharaa F, Abdalla AK (2009) Neurological complications of ankylosing spondylitis: neurophysiological assessment. Rheumatol Int 29:1031–1040
Lee W, Reveille JD, Weisman MH (2008) Women with ankylosing spondylitis: a review. Arthritis Rheum 59:449–454
Oh DH, Jun JB, Kim HT, Lee SW, Jung SS, Lee IH, Kim SY (2001) Transverse myelitis in a patient with long-standing ankylosing spondylitis. Clin Exp Rheumatol 19:195–196
Lan HH, Chen DY, Chen CC, Lan JL, Hsieh CW (2007) Combination of transverse myelitis and arachnoiditis in cauda equina syndrome of long-standing ankylosing spondylitis: MRI features and its role in clinical management. Clin Rheumatol 26:1963–1967
Mitchell MJ, Sartoris DJ, Moody D, Resnick D (1990) Cauda equina syndrome complicating ankylosing spondylitis. Radiology 175:521–525
Sofat N, Malik O, Higgens CS (2006) Neurological involvement in patients with rheumatic disease. QJM 99:69–79
Sá MJ Acute transverse myelitis: a practical reappraisal. Autoimmun Rev (in press)
Shimizu F, Kawai M, Koga M, Sano Y, Negoro K, Kanda T (2007) A forty-five-years-old woman suffered from Sjögren syndrome with progressive tetraparesis [Jpn]. Rinsho Shinkeigaku 47:502–506
Delalande S, de Seze J, Fauchais AL, Hachulla E, Stojkovic T, Ferriby D, Dubucquoi S, Pruvo JP, Vermersch P, Hatron PY (2004) Neurologic manifestations in primary Sjögren syndrome: a study of 82 patients. Medicine (Baltimore) 83:280–291
Attout H, Rahmeh F, Ziegler F (2000) de Gorgerot-Sjogren syndrome simulating amyotrophic lateral sclerosis [Fre]. Rev Med Interne 21:708–710
Schady W, Metcalfe RA, Holt PJ (1989) Rheumatoid arthritis and motor neurone disease—an association? Br J Rheumatol 28:70–73
Sostarko M, Brzovic Z, Vranjes D (1994) Motor neurone disease associated with several immunological disorders: a case report. J Neurol Sci 124(Suppl):70–71
Cereda C, Baiocchi C, Bongioanni P, Cova E, Guareschi S, Metelli MR, Rossi B, Sbalsi I, Cuccia MC, Ceroni M (2008) TNF and sTNFR1/2 plasma levels in ALS patients. J Neuroimmunol 194:123–131
Poloni M, Facchetti D, Mai R, Micheli A, Agnoletti L, Francolini G, Mora G, Camana C, Mazzini L, Bachetti T (2000) Circulating levels of tumor necrosis factor-alpha and its soluble receptors are increased in the blood of patients with amyotrophic lateral sclerosis. Neurosci Lett 287:211–214
Goncalves DU, Proietti FA, Barbosa-Stancioli EF, Martins ML, Ribas JG, Martins-Filho OA, Teixeira-Carvalho A, Peruhype-Magalhães V, Carneiro-Proietti AB (2008) HTLV-1-associated myelopathy/tropical spastic paraparesis (HAM/TSP) inflammatory network. Inflamm Allergy Drug Targets 7:98–107
Reveille JD, Arnett FC (2005) Spondyloarthritis: update on pathogenesis and management. Am J Med 118:592–603
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Chen, WH., Yin, HL., Lin, HS. et al. Symptomatic noncompressive motoromyelopathy presents as early manifestation in ankylosing spondylitis. Rheumatol Int 31, 945–950 (2011). https://doi.org/10.1007/s00296-010-1614-z
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-010-1614-z