Treatment-resistant CIDP in an IgG Tubulin Autoantibody Positive Patient
case report and review of the literature
DOI:
https://doi.org/10.17161/rrnmf.v1i5.14547Keywords:
CIDP, IgG tubulin autoantibodies, rituximab, demyelinating neuropathyAbstract
Objectives: To describe a case of rapidly relapsing chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) in the setting of positive serum IgG tubulin autoantibodies.
Methods: We wrote a case report and performed a literature review of IgG tubulin autoantibodies and the use of rituximab in treatment resistant CIDP.
Results: Our case report describes a 29-year-old woman with CIDP that was resistant to treatment with steroids, intravenous immunoglobulin, and plasma exchange. An extensive workup of her rapidly relapsing CIDP was negative, with the exception of positive serum IgG tubulin autoantibodies. She ultimately stabilized on oral steroids, plasma exchange and rituximab, with a regular recurrence of weakness occurring approximately every month that led to rehospitalization.
Conclusion: Anti-tubulin antibodies could be a marker of a subtype of CIDP that is treatment resistant. We detail her clinical course to serve as an example for other cases of IgG tubulin autoantibody positive CIDP patients that could be described in the future.
Metrics
Downloads
References
2 Schaik IN, Vermeulen M, Doom PA, Brand A. Anti-tubulin antibodies have no diagnostic value in patients with chronic inflammatory demyelinating polyneuropathy. Journal of Neurology 1995;242(9):599-603. doi:10.1007/bf00868814. PMID 8551323
3 Manfredini E, Nobile-Orazio E, Allaria S, Scarlato G. Anti-alpha- and beta-tubulin IgM antibodies in dysimmune neuropathies. Journal of the Neurological Sciences. 1995;133(1-2):79-84. doi:10.1016/0022-510x(95)00149-v. PMID 8583236
4 Kleyweg RP, van der Meché FG, Schmitz PI. Interobserver agreement in the assessment of muscle strength and functional abilities in Guillain-Barré syndrome. Muscle Nerve. 1991;14(11):1103-1109. Doi:10.1002/mus.880141111 PMID 1745285
5 European Federation of Neurological Societies/Peripheral Nerve Society Guideline on management of chronic inflammatory demyelinating polyradiculoneuropathy: Report of a joint task force of the European Federation of Neurological Societies and the Peripheral Nerve Society - First Revision. Journal of the Peripheral Nervous System 2010;15(1):1-9. doi:10.1111/j.1529-8027.2010.00245.x.
6 Bharath V, Eckert K, Kang M, Chin-Yee IH, Hsia CC. Incidence and natural history of intravenous immunoglobulin-induced aseptic meningitis: a retrospective review at a single tertiary care center. Transfusion 2015;55(11):2597-2605. doi:10.1111/trf.13200 PMID 26095012
7 Lucke IM, Peric S, Lieverloo GGAV, Wieske L, Verhamme C, Schaik IN, et al. Elevated leukocyte count in cerebrospinal fluid of patients with chronic inflammatory demyelinating polyneuropathy. Journal of the Peripheral Nervous System 2018;23(1):49-54. doi:10.1111/jns.12250. PMID 29341326
8 Roux T, Debs R, Maisonobe T, Lenglet T, Delorme C, Louapre C, et al. Rituximab in chronic inflammatory demyelinating polyradiculoneuropathy with associated diseases. Journal of the Peripheral Nervous System 2018;23(4):235-240. doi:10.1111/jns.12287. PMID 30203907
9 Benedetti L, Briani C, Franciotta D, Fazio R, Paolasso I, Comi C, et al. Rituximab in patients with chronic inflammatory demyelinating polyradiculoneuropathy: a report of 13 cases and review of the literature. Journal of Neurology, Neurosurgery & Psychiatry 2010;82(3):306-308. doi:10.1136/jnnp.2009.188912. PMID 20639381
10 Velardo D, Riva N, Carro UD, Bianchi F, Comi G, Fazio R. Rituximab in refractory chronic inflammatory demyelinating polyradiculoneuropathy: report of four cases. Journal of Neurology 2017;264(5):1011-1014. doi:10.1007/s00415-017-8462-7 PMID 28337614
11 Querol L, Rojas-García R, Diaz-Manera J, Barcena J, Pardo J, Ortega-Moreno A, et al. Rituximab in treatment-resistant CIDP with antibodies against paranodal proteins. Neurology - Neuroimmunology Neuroinflammation 2015;2(5). doi:10.1212/nxi.0000000000000149. PMID 26401517
12 Stubbs E, Fisher M, Wilson J, Siegel GJ. High-titer immunoglobulin M antibody to nerve-specific class III beta-tubulin in the serum of a patient with sensory demyelinating polyneuropathy. Muscle & Nerve December 1996:1638-1639. PMID 8941286
13 Connolly AM, Pestronk A. Anti‐Tubulin Autoantibodies in Acquired Demyelinating Polyneuropathies. The Journal of Infectious Diseases 1997;176(s2). doi:10.1086/513801. PMID 9396702
Downloads
Published
Issue
Section
License
Copyright (c) 2020 Alaina Giacobbe, Mitesh Patel, Scott Heller, Miguel Chuquilin
This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.
