Acute transverse myelitis revealing ankylosing spondylitis: A case report and literature review

Boussaid, S.; Rahmouni, S.; Rekik, S.; Jammali, Samia; Chéour, E.; Sahli, Hela; Elleuch, M.

doi:10.1002/ccr3.4878

Clinical Case Reports

2021

DOI: 10.1002/ccr3.4878

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Acute transverse myelitis revealing ankylosing spondylitis: A case report and literature review

S. Boussaid

S. Rahmouni

S. Rekik

et al.

Abstract: When faced with a patient with acute myelopathy, thorough investigations should be undertaken to determine the cause. Ankylosing spondylitis should be kept in mind as a possible cause.

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Cited by 3 publications

(6 citation statements)

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“… 12 , 16 , 17 Acute transverse myelitis can occur as a subset of several primary demyelinating disorders of the CNS (i.e., multiple sclerosis, NMOSD, myelin oligodendrocyte glycoprotein antibody disease, and acute disseminated encephalomyelitis) and various systemic autoimmune connective tissue disorders (i.e., systemic lupus erythematosus, mixed connective tissue disease, Sjögren syndrome, inflammatory bowel disease, and neurosarcoidosis). 18 Acute transverse myelitis (short or long segment) is an infrequent extra-articular complication of AS. 18 It has been reported to evolve either as a distinct neurological complication of AS, or it may develop secondary to TNF-alpha-inhibitor therapy for the treatment of AS.…”

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“… 18 Acute transverse myelitis (short or long segment) is an infrequent extra-articular complication of AS. 18 It has been reported to evolve either as a distinct neurological complication of AS, or it may develop secondary to TNF-alpha-inhibitor therapy for the treatment of AS. 18 , 19 AS is a heritable inflammatory spondyloarthropathy that primarily affects the axial skeleton, which is mediated by T-cells; B-cells only play a minor role.…”

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confidence: 99%

“… 18 It has been reported to evolve either as a distinct neurological complication of AS, or it may develop secondary to TNF-alpha-inhibitor therapy for the treatment of AS. 18 , 19 AS is a heritable inflammatory spondyloarthropathy that primarily affects the axial skeleton, which is mediated by T-cells; B-cells only play a minor role. 5 On the contrary, the key for the pathogenesis of NMOSD is the production of autoantibodies against AQP-4 channels expressed on astrocytes, leading to complement-mediated damage, with ensuing demyelination.…”

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confidence: 99%

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“… 18 It has been reported to evolve either as a distinct neurological complication of AS, or it may develop secondary to TNF-alpha-inhibitor therapy for the treatment of AS. 18 , 19…”

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confidence: 99%

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Seropositive Neuromyelitis Optica in a Case of Undiagnosed Ankylosing Spondylitis: A Neuro-Rheumatological Conundrum

Ghosh

Roy

León-Ruiz

et al. 2022

Qatar Medical Journal

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Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune astrocytopathy against foot processes of aquaporin-4 (AQP4) water channels. Patients with NMOSD tend to have other coexisting autoimmune/connective tissue diseases. However, AQP-4-antibody-positive NMOSD coexisting with ankylosing spondylitis (AS) is rare. AS is an immune-mediated disorder, a subset of axial spondyloarthropathies, which commonly manifests as chronic inflammatory back pain in young people, and it has a strong association with HLA-B27. In this study, a 35-year-old Indian man with an undiagnosed progressive axial spondyloarthropathy (i.e., AS) is reported presenting with acute-onset longitudinally extensive transverse myelitis, a clinical subset of NMOSD. Neuromyelitis optica spectrum disorder (NMOSD), a primary demyelinating disorder of the central nervous system (CNS), is an autoimmune astrocytopathy against foot processes of aquaporin-4 (AQP4) water channels, which manifests with optic neuritis, longitudinally extensive transverse myelitis (LETM), area-postrema syndrome, brainstem syndrome diencephalic syndrome, and cerebral syndrome. 1 – 4 Ankylosing spondylitis (AS) is an immune-mediated disorder, a subset of axial spondyloarthropathies, which commonly manifests as chronic inflammatory back pain in young people, and it has a strong association with HLA-B27. 5 , 6 AS characteristically targets the axial skeleton, peripheral joints, entheses (connective tissues between tendons/ligaments and bones), and gut. 5 , 6 Patients with NMOSD tend to have other coexisting autoimmune/connective tissue diseases. 7 For example, cases with NMOSD and multiple sclerosis, which are other autoimmune primary demyelinating disorders of the CNS, have been reported. 8 , 9 However, concurrent existence of AS and NMOSD in the same patient even over years of disease course is rare. 10 , 11 In addition, studies describing neurological manifestations of AS are limited, 12 and they focus on joint inflammation and long-standing bony pathology (ankylosis) related to compressive myelopathy, myelo-radiculopathy, and cauda equina syndromes. 12 , 13 The authors present a case of a young Indian man with an undiagnosed progressive AS (misdiagnosed and mismanaged by an indigenous medical practitioner) presenting with acute-onset LETM variant of AQP4-positive NMOSD. A 35-year-old healthy, non-comorbid man from rural India came to the outpatient department with complaints of persistent tingling, numbness, and weakness of both lower limbs (right more than left) for 10 days. The clinical picture showed acute-onset urinary retention, which was relieved by urinary cathete...

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confidence: 99%

mentioning

confidence: 99%

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confidence: 99%

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confidence: 99%

“… 18 It has been reported to evolve either as a distinct neurological complication of AS, or it may develop secondary to TNF-alpha-inhibitor therapy for the treatment of AS. 18 , 19…”

mentioning

confidence: 99%

See 3 more Smart Citations

Seropositive Neuromyelitis Optica in a Case of Undiagnosed Ankylosing Spondylitis: A Neuro-Rheumatological Conundrum

Ghosh

Roy

León-Ruiz

et al. 2022

Qatar Medical Journal

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Exposure to specific tumour necrosis factor inhibitors and risk of demyelinating and inflammatory neuropathy in cohorts of patients with inflammatory arthritis: a collaborative observational study across five Nordic rheumatology registers

et al. 2023

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ObjectiveTo compare incidences of neuroinflammatory events, including demyelinating disease (DML), inflammatory polyneuropathies (IPN) and multiple sclerosis (MS), in patients with rheumatoid arthritis (RA) or spondyloarthritis (SpA; including psoriatic arthritis) starting a tumour necrosis factor inhibitor (TNFi), investigating whether monoclonal TNFi antibodies (other TNFis (oTNFis)) confer higher risk than etanercept.MethodsThis is an observational cohort study including patients from the five Nordic countries starting a TNFi in 2001–2020. Time to first neuroinflammatory event was identified through register linkages. We calculated crude incidence rates (cIR) per 1000 person-years and used multivariable-adjusted Cox regression to compare incidences of neuroinflammatory events overall and for DML, IPN and MS with oTNFi versus etanercept. We further examined individual TNFis and indications.Results33 883 patients with RA and 28 772 patients with SpA were included, initiating 52 704 and 46 572 treatment courses, respectively. In RA, we observed 135 neuroinflammatory events (65% DML) with cIR of 0.38 with oTNFi and 0.34 with etanercept. The HR of oTNFi versus etanercept was 1.07 (95% CI 0.74 to 1.54) for any neuroinflammatory event, 0.79 (95% CI 0.51 to 1.22) for DML, 2.20 (95% CI 1.05 to 4.63) for IPN and 0.73 (95% CI 0.34 to 1.56) for MS. In SpA, we observed 179 events (78% DML) with cIR of 0.68 with oTNFi and 0.65 with etanercept. The HR for any neuroinflammatory event, DML, IPN and MS was 1.06 (95% CI 0.75 to 1.50), 1.01 (95% CI 0.68 to 1.50), 1.28 (95% CI 0.61 to 2.69) and 0.94 (95% CI0.53 to 1.69), respectively.ConclusionThe cIRs of neuroinflammatory events are higher in SpA than in RA, but the choice of specific TNFi does not seem to play an important role in the risk of neuroinflammatory events.

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Bilateral lower limb weakness due to transverse myelitis as a first presentation of ankylosing spondylitis

Salim,

Abuhashem,

Nassar

et al. 2024

Arab Journal of Rheumatology

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The occurrence of transverse myelitis (TM) in ankylosing spondylitis (AS) patients is a rarely reported event with only a few reports of similar cases in existing literature. When faced with a patient with TM, attention should be focused on determining the cause. AS should be kept in mind as a possible etiology. We report the case of a 58-year-old female patient who presented at Al-Ahli Hospital in Palestine, with a 3-day history of lower limb weakness, preceded by a 1-month history of lower back pain with radiation to lower limbs. Based on clinical findings and a comprehensive diagnostic work-up, the patient was diagnosed with TM along with AS. Steroid therapy was started, with pulse therapy having minimal effect. Due to the poor response to steroid therapy, plasmapheresis was done with a good response. After careful consideration, our patient was discharged on low-dose oral glucocorticoids for her TM, and methotrexate for her AS, with close follow-up by a neurologist and a rheumatologist.

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Acute transverse myelitis revealing ankylosing spondylitis: A case report and literature review

Abstract: When faced with a patient with acute myelopathy, thorough investigations should be undertaken to determine the cause. Ankylosing spondylitis should be kept in mind as a possible cause.

Cited by 3 publications

References 29 publications

Seropositive Neuromyelitis Optica in a Case of Undiagnosed Ankylosing Spondylitis: A Neuro-Rheumatological Conundrum

Seropositive Neuromyelitis Optica in a Case of Undiagnosed Ankylosing Spondylitis: A Neuro-Rheumatological Conundrum

Exposure to specific tumour necrosis factor inhibitors and risk of demyelinating and inflammatory neuropathy in cohorts of patients with inflammatory arthritis: a collaborative observational study across five Nordic rheumatology registers

Bilateral lower limb weakness due to transverse myelitis as a first presentation of ankylosing spondylitis

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