Influenza-associated MOG antibody-positive longitudinally extensive transverse myelitis: a case report

BMC Neurology, Nov 2014

Background Myelin-oligodendrocyte glycoprotein antibody (MOG antibodies) was found in various demyelinated diseases. This is the first report of a patient with longitudinally extensive transverse myelitis with an extremely high titer of MOG antibodies after an influenza infection. This case supports the view that MOG antibodies are linked to longitudinally extensive transverse myelitis and that influenza infection might trigger the MOG antibodies. Case presentation A 32-year-old healthy male developed high fever, dysesthesia and paraesthesia below the C2 area, muscle weakness of the bilateral lower extremities, and urinary retention ten days after an influenza type A infection. Magnetic resonance imaging revealed a longitudinal lesion in the spinal cord extending from C2 to the spinal conus. There were no lesions in the brain or optic nerves. Established cell-based immunoassays revealed that he was positive for MOG antibodies (titer = 65,536) and negative for anti-aquaporin 4 antibodies (AQP4 antibodies). He fully recovered after steroid pulse therapy followed by 60 mg prednisolone. Conclusion This is the first report of influenza A-associated longitudinally extensive transverse myelitis with a high titer anti-MOG antibodies. Our case report supports a relationship between anti-MOG antibodies and longitudinally extensive transverse myelitis, which was triggered by influenza infection. Further studies are needed to establish the clinical significance of anti-MOG antibodies for diagnosis, treatment, and prognosis.

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Influenza-associated MOG antibody-positive longitudinally extensive transverse myelitis: a case report

BMC Neurology Influenza-associated MOG antibody-positive longitudinally extensive transverse myelitis: a case report Haruka Amano 0 Nobukazu Miyamoto 0 Hideki Shimura 0 Douglas Kazutoshi Sato 1 Kazuo Fujihara 1 Shinichi Ueno 0 Ryota Nakamura 0 Yuji Ueno 0 Masao Watanabe 0 Nobutaka Hattori 2 Takao Urabe 0 0 Department of Neurology, Juntendo University Urayasu Hospital , 2-1-1 Tomioka, Urayasu, Chiba 279-0021 , Japan 1 Department of Neurology, Tohoku University Graduate School of Medicine , Miyagi , Japan 2 Department of Neurology, Juntendo University School of Medicine , Tokyo , Japan Background: Myelin-oligodendrocyte glycoprotein antibody (MOG antibodies) was found in various demyelinated diseases. This is the first report of a patient with longitudinally extensive transverse myelitis with an extremely high titer of MOG antibodies after an influenza infection. This case supports the view that MOG antibodies are linked to longitudinally extensive transverse myelitis and that influenza infection might trigger the MOG antibodies. Case presentation: A 32-year-old healthy male developed high fever, dysesthesia and paraesthesia below the C2 area, muscle weakness of the bilateral lower extremities, and urinary retention ten days after an influenza type A infection. Magnetic resonance imaging revealed a longitudinal lesion in the spinal cord extending from C2 to the spinal conus. There were no lesions in the brain or optic nerves. Established cell-based immunoassays revealed that he was positive for MOG antibodies (titer = 65,536) and negative for anti-aquaporin 4 antibodies (AQP4 antibodies). He fully recovered after steroid pulse therapy followed by 60 mg prednisolone. Conclusion: This is the first report of influenza A-associated longitudinally extensive transverse myelitis with a high titer anti-MOG antibodies. Our case report supports a relationship between anti-MOG antibodies and longitudinally extensive transverse myelitis, which was triggered by influenza infection. Further studies are needed to establish the clinical significance of anti-MOG antibodies for diagnosis, treatment, and prognosis. Myelin-oligodendrocyte glycoprotein antibody; Longitudinal extensive transverse myelitis; Neuromyelitis optica; Aquaporin-4 antibody Background Myelin oligodendrocyte glycoprotein (MOG) localizes on the outermost surface of the myelin sheath and oligodendrocytes in the central nervous system (CNS). Autoantibodies against MOG are reportedly found in patients with a spectrum of inflammatory demyelinating diseases of the CNS, including acute disseminated encephalomyelitis, multiple sclerosis, transverse myelitis, and neuromyelitis optica (NMO) [ 1 ]. NMO is a severe inflammatory disorder of the central nervous system that predominantly affects the optic nerves and spinal cord. Most patients with NMO have antibodies against the water channel aquaporin-4 (AQP4 antibodies), which are thought to be pathogenic [ 2-4 ]. However, some patients are seronegative for AQP4 antibody; the lack of a biomarker makes diagnosis and management of these patients difficult. Many clinicians perceive these patients to be similar to those with AQP4-Abs and manage them in the same way. Recently, some groups showed that some AQP4 antibody seronegative patients have antibodies against MOG [ 5-7 ]. They also demonstrated differences in clinical phenotypes and responsiveness of therapy between the AQP4 antibody seronegative and MOG antibody seropositive patients with NMO/NMO spectrum disorder (NMOSD). Detection of MOG antibodies is important for patients management. Mice that are transgenic for MOG-specific T-cell and B-cell receptors develop spontaneous experimental autoimmune encephalomyelitis (EAE) [ 8-11 ]. Recent studies suggest that MOG-related EAE can mimic a neurological syndrome closely resembling NMO. Although the clinical spectrum of MOG IgG associated diseases in humans is reflected in different experimental models, the role of MOG antibodies in pathogenesis is still unclear. Here we describe a patient who suffered from longitudinal extended TM (LETM) with high-titer MOG antibodies following an influenza-A infection and his AQP4 antibody was negative. Case presentation A 32-year-old male, without any relevant medical history, felt general fatigue and had a high fever of 38.9 degrees Celsius. The following day, he was diagnosed with influenza type A by nasal swab test at a clinic, and prescribed oseltamivir. His symptoms were ameliorated sufficiently that he was able to return to work from day 5 and go winter climbing on days 6–7. On day 9, however, he experienced whole body pain, urinary retention, and weakness of the bilateral lower extremities; these symptoms then became exacerbated. Eventually, he was unable to walk by himself, and came to our hospital on day 10. His body temperature was 39.4 degrees Celsius and there was marked bilateral lower extremity weakness. Deep tendon reflexes were normal in both the upper an (...truncated)


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Haruka Amano, Nobukazu Miyamoto, Hideki Shimura, Douglas Sato, Kazuo Fujihara, Shinichi Ueno, Ryota Nakamura, Yuji Ueno, Masao Watanabe, Nobutaka Hattori, Takao Urabe. Influenza-associated MOG antibody-positive longitudinally extensive transverse myelitis: a case report, BMC Neurology, 2014, pp. 224, 14, DOI: 10.1186/s12883-014-0224-x