Abstract
OBJECTIVES—The effect of interferon-β1a (INF-β1a; Rebif®) was studied in patients with chronic motor neuropathies not improving after conventional treatments such as immunoglobulins, steroids, cyclophosphamide or plasma exchange. METHODS—A prospective open study was performed with a duration of 6-12 months. Three patients with a multifocal motor neuropathy and one patient with a pure motor form of chronic inflammatory demyelinating polyneuropathy were enrolled. Three patients had anti-GM1 antibodies. Treatment consisted of subcutaneous injections of IBF-β1a (6 MIU), three times a week. Primary outcome was assessed at the level of disability using the nine hole peg test, the 10 metres walking test, and the modified Rankin scale. Secondary outcome was measured at the impairment level using a slightly modified MRC sumscore. RESULTS—All patients showed a significant improvement on the modified MRC sumscore. The time required to walk 10 metres and to fulfil the nine hole peg test was also significantly reduced in the first 3 months in most patients. However, the translation of these results to functional improvement on the modified Rankin was only seen in two patients. There were no severe adverse events. Motor conduction blocks were partially restored in one patient only. Anti-GM1 antibody titres did not change. CONCLUSION—These findings indicate that severely affected patients with chronic motor neuropathies not responding to conventional therapies may improve when treated with INF-β1a. From this study it is suggested that INF-β1a should be administered in patients with chronic motor neuropathies for a period of up to 3 months before deciding to cease treatment. A controlled trial is necessary to confirm these findings.
Full Text
The Full Text of this article is available as a PDF (143.7 KB).
Selected References
These references are in PubMed. This may not be the complete list of references from this article.
- Chaudhry V., Corse A. M., Cornblath D. R., Kuncl R. W., Drachman D. B., Freimer M. L., Miller R. G., Griffin J. W. Multifocal motor neuropathy: response to human immune globulin. Ann Neurol. 1993 Mar;33(3):237–242. doi: 10.1002/ana.410330303. [DOI] [PubMed] [Google Scholar]
- Choudhary P. P., Thompson N., Hughes R. A. Improvement following interferon beta in chronic inflammatory demyelinating polyradiculoneuropathy. J Neurol. 1995 Mar;242(4):252–253. doi: 10.1007/BF00919601. [DOI] [PubMed] [Google Scholar]
- Collen F. M., Wade D. T., Bradshaw C. M. Mobility after stroke: reliability of measures of impairment and disability. Int Disabil Stud. 1990 Jan-Mar;12(1):6–9. doi: 10.3109/03790799009166594. [DOI] [PubMed] [Google Scholar]
- Feldman E. L., Bromberg M. B., Albers J. W., Pestronk A. Immunosuppressive treatment in multifocal motor neuropathy. Ann Neurol. 1991 Sep;30(3):397–401. doi: 10.1002/ana.410300312. [DOI] [PubMed] [Google Scholar]
- Gorson K. C., Ropper A. H., Clark B. D., Dew R. B., 3rd, Simovic D., Allam G. Treatment of chronic inflammatory demyelinating polyneuropathy with interferon-alpha 2a. Neurology. 1998 Jan;50(1):84–87. doi: 10.1212/wnl.50.1.84. [DOI] [PubMed] [Google Scholar]
- Jacobs B. C., van Doorn P. A., Schmitz P. I., Tio-Gillen A. P., Herbrink P., Visser L. H., Hooijkass H., van der Meché F. G. Campylobacter jejuni infections and anti-GM1 antibodies in Guillain-Barré syndrome. Ann Neurol. 1996 Aug;40(2):181–187. doi: 10.1002/ana.410400209. [DOI] [PubMed] [Google Scholar]
- Kleyweg R. P., van der Meché F. G., Schmitz P. I. Interobserver agreement in the assessment of muscle strength and functional abilities in Guillain-Barré syndrome. Muscle Nerve. 1991 Nov;14(11):1103–1109. doi: 10.1002/mus.880141111. [DOI] [PubMed] [Google Scholar]
- Krendel D. A. Biopsy findings link multifocal motor neuropathy to chronic inflammatory demyelinating polyneuropathy. Ann Neurol. 1996 Dec;40(6):948–950. doi: 10.1002/ana.410400626. [DOI] [PubMed] [Google Scholar]
- Mariette X., Chastang C., Clavelou P., Louboutin J. P., Leger J. M., Brouet J. C. A randomised clinical trial comparing interferon-alpha and intravenous immunoglobulin in polyneuropathy associated with monoclonal IgM. The IgM-associated Polyneuropathy Study Group. J Neurol Neurosurg Psychiatry. 1997 Jul;63(1):28–34. doi: 10.1136/jnnp.63.1.28. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Noronha A., Toscas A., Jensen M. A. Interferon beta decreases T cell activation and interferon gamma production in multiple sclerosis. J Neuroimmunol. 1993 Jul;46(1-2):145–153. doi: 10.1016/0165-5728(93)90244-s. [DOI] [PubMed] [Google Scholar]
- Oh S. J., Claussen G. C., Odabasi Z., Palmer C. P. Multifocal demyelinating motor neuropathy: pathologic evidence of 'inflammatory demyelinating polyradiculoneuropathy'. Neurology. 1995 Oct;45(10):1828–1832. doi: 10.1212/wnl.45.10.1828. [DOI] [PubMed] [Google Scholar]
- Otto M., Stein H., Szudra A., Zerr I., Bodemer M., Gefeller O., Poser S., Kretzschmar H. A., Mäder M., Weber T. S-100 protein concentration in the cerebrospinal fluid of patients with Creutzfeldt-Jakob disease. J Neurol. 1997 Sep;244(9):566–570. doi: 10.1007/s004150050145. [DOI] [PubMed] [Google Scholar]
- Pestronk A., Cornblath D. R., Ilyas A. A., Baba H., Quarles R. H., Griffin J. W., Alderson K., Adams R. N. A treatable multifocal motor neuropathy with antibodies to GM1 ganglioside. Ann Neurol. 1988 Jul;24(1):73–78. doi: 10.1002/ana.410240113. [DOI] [PubMed] [Google Scholar]
- Pollard J. D., McCombe P. A., Baverstock J., Gatenby P. A., McLeod J. G. Class II antigen expression and T lymphocyte subsets in chronic inflammatory demyelinating polyneuropathy. J Neuroimmunol. 1986 Dec;13(2):123–134. doi: 10.1016/0165-5728(86)90059-7. [DOI] [PubMed] [Google Scholar]
- Sabatelli M., Mignogna T., Lippi G., Milone M., Di Lazzaro V., Tonali P., Bertini E. Interferon-alpha may benefit steroid unresponsive chronic inflammatory demyelinating polyneuropathy. J Neurol Neurosurg Psychiatry. 1995 May;58(5):638–639. doi: 10.1136/jnnp.58.5.638. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Thompson N., Choudhary P., Hughes R. A., Quinlivan R. M. A novel trial design to study the effect of intravenous immunoglobulin in chronic inflammatory demyelinating polyradiculoneuropathy. J Neurol. 1996 Mar;243(3):280–285. doi: 10.1007/BF00868527. [DOI] [PubMed] [Google Scholar]
- Van den Berg L. H., Kerkhoff H., Oey P. L., Franssen H., Mollee I., Vermeulen M., Jennekens F. G., Wokke J. H. Treatment of multifocal motor neuropathy with high dose intravenous immunoglobulins: a double blind, placebo controlled study. J Neurol Neurosurg Psychiatry. 1995 Sep;59(3):248–252. doi: 10.1136/jnnp.59.3.248. [DOI] [PMC free article] [PubMed] [Google Scholar]
- Weinstock-Guttman B., Ransohoff R. M., Kinkel R. P., Rudick R. A. The interferons: biological effects, mechanisms of action, and use in multiple sclerosis. Ann Neurol. 1995 Jan;37(1):7–15. doi: 10.1002/ana.410370105. [DOI] [PubMed] [Google Scholar]
- van Doorn P. A., Brand A., Strengers P. F., Meulstee J., Vermeulen M. High-dose intravenous immunoglobulin treatment in chronic inflammatory demyelinating polyneuropathy: a double-blind, placebo-controlled, crossover study. Neurology. 1990 Feb;40(2):209–212. doi: 10.1212/wnl.40.2.209. [DOI] [PubMed] [Google Scholar]
- van Swieten J. C., Koudstaal P. J., Visser M. C., Schouten H. J., van Gijn J. Interobserver agreement for the assessment of handicap in stroke patients. Stroke. 1988 May;19(5):604–607. doi: 10.1161/01.str.19.5.604. [DOI] [PubMed] [Google Scholar]