Widening of myelin lamellae in polyneuropathy with immunoglobulin-M monoclonal gammopathy, without activity against myelin-associated glycoprotein, responsive to treatment.


Journal

Neuromuscular disorders : NMD
ISSN: 1873-2364
Titre abrégé: Neuromuscul Disord
Pays: England
ID NLM: 9111470

Informations de publication

Date de publication:
08 2022
Historique:
received: 07 03 2022
revised: 04 05 2022
accepted: 09 05 2022
pubmed: 27 5 2022
medline: 19 8 2022
entrez: 26 5 2022
Statut: ppublish

Résumé

We report the case of a patient with a very severe predominantly demyelinating sensorimotor polyneuropathy (with axonal loss) that had developed over several months, along with an immunoglobulin-M monoclonal gammopathy without anti-myelin associated glycoprotein antibodies (or other antibodies against myelin). Widening of myelin lamellae were frequently observed by electron microscopic examination of a nerve biopsy: immunoglobulin-M targeting an unknown myelin antigen appears to be responsible for the nerve lesions similar to those observed in anti-myelin associated glycoprotein polyneuropathy. Usually, if in anti-myelin associated glycoprotein neuropathy the response to immunotherapies is not optimal, in this case the combination of plasma exchanges and rituximab was effective, suggesting an autoimmune origin.

Identifiants

pubmed: 35618575
pii: S0960-8966(22)00138-9
doi: 10.1016/j.nmd.2022.05.001
pii:
doi:

Substances chimiques

Antibodies, Monoclonal 0
Immunoglobulin M 0
Myelin-Associated Glycoprotein 0

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

678-681

Informations de copyright

Copyright © 2022. Published by Elsevier B.V.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors declare that they have no conflict of interest.

Auteurs

Jean-Michel Vallat (JM)

Department of Neurology, National Reference Center for 'Rare Peripheral Neuropathies', University Hospital of Limoges (CHU Limoges - Dupuytren Hospital), 2 avenue Martin Luther King, Limoges 87042, France.

Nathalie Deschamps (N)

Department of Neurology, National Reference Center for 'Rare Peripheral Neuropathies', University Hospital of Limoges (CHU Limoges - Dupuytren Hospital), 2 avenue Martin Luther King, Limoges 87042, France.

Laurence Richard (L)

Department of Neurology, National Reference Center for 'Rare Peripheral Neuropathies', University Hospital of Limoges (CHU Limoges - Dupuytren Hospital), 2 avenue Martin Luther King, Limoges 87042, France.

Laurent Magy (L)

Department of Neurology, National Reference Center for 'Rare Peripheral Neuropathies', University Hospital of Limoges (CHU Limoges - Dupuytren Hospital), 2 avenue Martin Luther King, Limoges 87042, France.

Jérôme Devaux (J)

Institute for Neurosciences of Montpellier, INSERM U1051, Montpellier University, Montpellier, France.

Stéphane Mathis (S)

Department of Neurology, Nerve-Muscle Unit, AOC National Reference Center for Neuromuscular Disorders, ALS Center, University Hospital of Bordeaux (CHU Bordeaux - Pellegrin Hospital), place Amélie Raba-Léon, Bordeaux 33000, France. Electronic address: stephane.mathis@chu-bordeaux.fr.

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Classifications MeSH