Therapeutic effectiveness of thalidomide in a patient with treatment-resistant restless legs syndrome.


Journal

Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine
ISSN: 1550-9397
Titre abrégé: J Clin Sleep Med
Pays: United States
ID NLM: 101231977

Informations de publication

Date de publication:
15 10 2020
Historique:
pubmed: 31 7 2020
medline: 24 6 2021
entrez: 31 7 2020
Statut: ppublish

Résumé

Recent developments in the genetics of restless legs syndrome (RLS) revealed associations of disease risk with genetic loci containing the genes coding cereblon, the protein bound by thalidomide, and its endogenous substrate MEIS2, whose degradation is inhibited by the thalidomide-cereblon interaction. Therefore it was hypothesized that thalidomide may be a potential treatment option for RLS. Here we report on the therapeutic effect of thalidomide in a patient with otherwise treatment-resistant RLS who received 100 mg thalidomide off-label for 3 weeks. The female patient, severely affected by RLS before treatment, experienced significant amelioration of the symptoms, increased self-reported sleep quality, and better daytime functioning during thalidomide treatment. This therapeutic success warrants larger studies investigating the efficacy of drugs of the thalidomide class in RLS.

Identifiants

pubmed: 32729462
doi: 10.5664/jcsm.8696
pmc: PMC7954015
doi:

Substances chimiques

Thalidomide 4Z8R6ORS6L

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

1815-1817

Informations de copyright

© 2020 American Academy of Sleep Medicine.

Références

Sleep Med. 2016 Apr;20:116-22
pubmed: 27318235
Sleep. 1999 Feb 1;22(1):113-5
pubmed: 9989372
J Pharmacol Exp Ther. 1961 Oct;134:60-8
pubmed: 14460349
Recent Pat Endocr Metab Immune Drug Discov. 2011 Sep;5(3):192-6
pubmed: 21913886
Neurology. 2002 Dec 24;59(12):1872-5
pubmed: 12499476
Sleep. 1991 Dec;14(6):540-5
pubmed: 1798888
Lancet Neurol. 2017 Nov;16(11):898-907
pubmed: 29029846
Nature. 2014 Aug 7;512(7512):49-53
pubmed: 25043012
Biochem Biophys Res Commun. 2018 Dec 9;507(1-4):443-449
pubmed: 30458989
Birth Defects Res C Embryo Today. 2015 Jun;105(2):140-56
pubmed: 26043938
Sleep Med. 2003 Mar;4(2):121-32
pubmed: 14592342
Mov Disord. 2018 Jul;33(7):1077-1091
pubmed: 29756335

Auteurs

Aaro V Salminen (AV)

Institute of Neurogenomics, Helmholtz Zentrum München GmbH - German Research Center for Environmental Health, Neuherberg, Germany.
Institute of Human Genetics, Technische Universität München, Munich, Germany.
Department of Neurology, Philipps-Universität Marburg, Marburg, Germany.

Nathalie Schandra (N)

Institute of Neurogenomics, Helmholtz Zentrum München GmbH - German Research Center for Environmental Health, Neuherberg, Germany.
Department of Neurology, Philipps-Universität Marburg, Marburg, Germany.

Barbara Schormair (B)

Institute of Neurogenomics, Helmholtz Zentrum München GmbH - German Research Center for Environmental Health, Neuherberg, Germany.
Institute of Human Genetics, Technische Universität München, Munich, Germany.

Konrad Oexle (K)

Institute of Neurogenomics, Helmholtz Zentrum München GmbH - German Research Center for Environmental Health, Neuherberg, Germany.

Juliane Winkelmann (J)

Institute of Neurogenomics, Helmholtz Zentrum München GmbH - German Research Center for Environmental Health, Neuherberg, Germany.
Institute of Human Genetics, Technische Universität München, Munich, Germany.
Chair of Neurogenetics, Klinikum Rechts der Isar, Technische Universität München, Munich, Germany.
Munich Cluster for Systems Neurology (SyNergy), Munich, Germany.

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