Abstract
Previous case reports and studies have shown that anti-myelin-associated glycoprotein (MAG) antibody can be detected in patients with polyneuropathy without any detectable M-protein. Nevertheless, the frequency of and related factors have not yet been adequately investigated. The objectives of this study are to examine the prevalence of anti-MAG antibody in patients with demyelinating neuropathy without M-protein and to determine their clinical characteristics. From January, 2004, to September, 2016, consecutive patients with chronic demyelinating neuropathy were recruited. Anti-MAG antibody presence was tested at the first evaluation. We determined the prevalence of anti-MAG antibody without M-protein among included patients and evaluated the clinical characteristics. A total of 44 patients were included in the present study (12 women; median age at first visit 60 years [interquartile range 47–67 years]; median duration between onset and first visit 9 months [3–26 months]). M-protein was found in eight patients (18%) at the first evaluation. Anti-MAG antibody was present in 2 of remaining 36 (5.6 [95% confidence interval 0–13.0] %) patients without M-protein. Patients with anti-MAG antibody exhibited slowly progressive and distal dominant neuropathy with elevated serum IgM levels and refractory to immunotherapy. There were no differences in clinical features between patients having anti-MAG antibody without M-protein, and those with M-protein. One patient with the anti-MAG antibody showed a delayed appearance of M-protein during a 4-year follow-up after diagnosis. The prevalence of the anti-MAG antibody in chronic demyelinating neuropathy without any detectable M-protein was 5.6%. Anti-MAG antibody may be detectable earlier than M-protein.
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Ellie E, Vital A, Steck A, Boiron JM, Vital C, Julien J (1996) Neuropathy associated with “benign” anti-myelin-associated glycoprotein IgM gammopathy: clinical, immunological, neurophysiological pathological findings and response to treatment in 33 cases. J Neurol 243:34–43
Nobile-Orazio E, Manfredini E, Carpo M et al (1994) Frequency and clinical correlates of anti-neural IgM antibodies in neuropathy associated with IgM monoclonal gammopathy. Ann Neurol 36:416–424
Lunn MP, Nobile-Orazio E (2012) Immunotherapy for IgM anti-myelin-associated glycoprotein paraprotein-associated peripheral neuropathies. Cochrane Database Syst Rev:CD002827
Kuijf ML, Eurelings M, Tio-Gillen AP et al (2009) Detection of anti-MAG antibodies in polyneuropathy associated with IgM monoclonal gammopathy. Neurology 73:688–695
European Federation of Neurological Societies/Peripheral Nerve Society Guideline on management of paraproteinemic demyelinating neuropathies (2010) Report of a Joint Task Force of the European Federation of Neurological Societies and the Peripheral Nerve Society—first revision. J Peripher Nerv Syst 15:185–195
Eurelings M, Moons KG, Notermans NC et al (2001) Neuropathy and IgM M-proteins: prognostic value of antibodies to MAG, SGPG, and sulfatide. Neurology 56:228–233
Kawagashira Y, Koike H, Ohyama K et al (2015) Axonal loss influences the response to rituximab treatment in neuropathy associated with IgM monoclonal gammopathy with anti-myelin-associated glycoprotein antibody. J Neurol Sci 348:67–73
Leger JM, Viala K, Nicolas G et al (2013) Placebo-controlled trial of rituximab in IgM anti-myelin-associated glycoprotein neuropathy. Neurology 80:2217–2225
Lupu VD, Mora CA, Dambrosia J, Meer J, Dalakas M, Floeter MK (2007) Terminal latency index in neuropathy with antibodies against myelin-associated glycoproteins. Muscle Nerve 35:196–202
Gabriel JM, Erne B, Bernasconi L et al (1998) Confocal microscopic localization of anti-myelin-associated glycoprotein autoantibodies in a patient with peripheral neuropathy initially lacking a detectable IgM gammopathy. Acta Neuropathol 95:540–546
Nobile-Orazio E, Latov N, Hays AP et al (1984) Neuropathy and anti-MAG antibodies without detectable serum M-protein. Neurology 34:218–221
Katz JS, Saperstein DS, Gronseth G, Amato AA, Barohn RJ (2000) Distal acquired demyelinating symmetric neuropathy. Neurology 54:615–620
Tagawa Y, Yuki N, Hirata K (2000) Anti-SGPG antibody in CIDP: nosological position of IgM anti-MAG/SGPG antibody-associated neuropathy. Muscle Nerve 23:895–899
Van den Berg L, Hays AP, Nobile-Orazio E et al (1996) Anti-MAG and anti-SGPG antibodies in neuropathy. Muscle Nerve 19:637–643
Kaku DA, England JD, Sumner AJ (1994) Distal accentuation of conduction slowing in polyneuropathy associated with antibodies to myelin-associated glycoprotein and sulphated glucuronyl paragloboside. Brain 117(Pt 5):941–947
Nobile-Orazio E, Francomano E, Daverio R et al (1989) Anti-myelin-associated glycoprotein IgM antibody titers in neuropathy associated with macroglobulinemia. Ann Neurol 26:543–550
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Sakamoto, Y., Shimizu, T., Tobisawa, S. et al. Chronic demyelinating neuropathy with anti-myelin-associated glycoprotein antibody without any detectable M-protein. Neurol Sci 38, 2165–2169 (2017). https://doi.org/10.1007/s10072-017-3133-0
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DOI: https://doi.org/10.1007/s10072-017-3133-0