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    Alemtuzumab in the treatment of IVIG-dependent chronic inflammatory demyelinating polyneuropathy.

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    Authors
    Marsh, E A
    Hirst, C L
    Llewelyn, J G
    Cossburn, M D
    Reilly, M M
    Krishnan, A
    Doran, M
    Ryan, A M
    Coles, A J
    Jones, J L
    Robertson, N P
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    Affiliation
    Department of Neurology, University Hospital of Wales, Heath Park, Cardiff, CF14 4XN, UK.
    Issue Date
    2010-06
    MeSH
    Adolescent
    Adult
    Age of Onset
    Anemia, Hemolytic, Autoimmune
    Antibodies, Monoclonal
    Antibodies, Monoclonal, Humanized
    Antibodies, Neoplasm
    Child
    Drug Therapy, Combination
    Female
    Humans
    Immunoglobulins, Intravenous
    Immunologic Factors
    Male
    Middle Aged
    Polyradiculoneuropathy, Chronic Inflammatory Demyelinating
    Time Factors
    Treatment Outcome
    Young Adult
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    Citation
    Alemtuzumab in the treatment of IVIG-dependent chronic inflammatory demyelinating polyneuropathy. 2010, 257 (6):913-9 J. Neurol.
    Journal
    Journal of neurology
    URI
    http://hdl.handle.net/10147/200270
    DOI
    10.1007/s00415-009-5437-3
    PubMed ID
    20049473
    Abstract
    Chronic inflammatory demyelinating polyneuropathy (CIDP) is an idiopathic immune mediated neuropathy causing demyelination and conduction block thought to occur as the result of an aberrant autoimmune response resulting in peripheral nerve inflammation mediated by T cells and humoral factors. Diagnosis commonly prompts initial treatment with steroids or intravenous immunoglobulin (IVIG) on which 5-35% subsequently become dependent to maintain function. Despite a number of small scale trials, the role for alternative long-term immunosuppression remains unclear. Alemtuzumab is a humanised monoclonal antibody targeting the CD52 antigen present on the surface of lymphocytes and monocytes. A single intravenous infusion results in rapid and profound lymphopoenia lasting >12 months. We report its use and clinical outcome in a small series of patients with severe IVIG-dependent CIDP. Seven patients (4 Males; 3 Females) who had failed to respond to conventional immunosuppression were treated in 5 centres receiving 9 courses of alemtuzumab (dose range 60-150 mg). Following treatment, mean monthly IVIG use fell 26% from 202 to 149 g and IVIG administration frequency from 22 to 136 days. Two patients had prolonged remission, two patients had a partial response and no clear benefit was observed in the remaining three patients (2 Males, 1 Females). Responding patients had a younger age at onset (19.5 years) and shorter disease duration than non-responders. Three patients developed autoimmune disease following treatment. Alemtuzumab may offer an alternative treatment for a subset of early onset IVIG dependent CIDP patients failing conventional immunosuppressive agents, but concerns about toxicity may limit its use.
    Item Type
    Article
    Language
    en
    ISSN
    1432-1459
    ae974a485f413a2113503eed53cd6c53
    10.1007/s00415-009-5437-3
    Scopus Count
    Collections
    Cork University Hospital

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