Sustained response to subcutaneous immunoglobulins in chronic ataxic neuropathy with anti-disialosyl IgM antibodies (CANDA): report of two cases and review of the literature.
Anti-disialosyl antibodies
B-cell lymphoma
CANDA
Chronic ataxic neuropathy
Subcutaneous immunoglobulins
Therapy
Journal
Journal of neurology
ISSN: 1432-1459
Titre abrégé: J Neurol
Pays: Germany
ID NLM: 0423161
Informations de publication
Date de publication:
Aug 2020
Aug 2020
Historique:
received:
07
11
2019
accepted:
16
04
2020
revised:
14
04
2020
pubmed:
30
4
2020
medline:
22
6
2021
entrez:
30
4
2020
Statut:
ppublish
Résumé
Chronic ataxic neuropathy with anti-disialosyl IgM antibodies (CANDA) is a rare disorder for which the pathological, neurophysiological, and therapeutic evidence remains anecdotal and controversial. This report on CANDA focuses on the neurophysiological patterns and treatment responses shared by two cases. One patient underwent nerve ultrasound follow-up. A comprehensive review of the literature highlighted the diverse experiences with different treatment options. Response to different therapies was similar in both patients: intravenous immunoglobulins achieved a favorable response albeit with significant wearing-off fluctuations; treatment with subcutaneous immunoglobulins (SCIg) was an effective alternative leading to a clinical response for at least 2 years. Rituximab, which was trialed in both patients, was not continued long enough to determine its efficacy in modifying the disease course and/or modulating responsiveness to immunoglobulins. Steroids caused clinical worsening in both patients. Immunoglobulin therapy appeared as the most effective in the treatment of these two patients. SCIg provided an effective treatment option for the long-term management of CANDA.
Identifiants
pubmed: 32347337
doi: 10.1007/s00415-020-09843-y
pii: 10.1007/s00415-020-09843-y
doi:
Substances chimiques
Gangliosides
0
Immunoglobulin M
0
Immunoglobulins
0
Immunoglobulins, Intravenous
0
Types de publication
Case Reports
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
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