PMID- 22683336
OWN - NLM
STAT- MEDLINE
DCOM- 20121018
LR  - 20220311
IS  - 1872-8421 (Electronic)
IS  - 0165-5728 (Linking)
VI  - 250
IP  - 1-2
DP  - 2012 Sep 15
TI  - Anti-MuSK- and anti-AChR-positive myasthenia gravis induced by d-penicillamine.
PG  - 94-8
LID - 10.1016/j.jneuroim.2012.05.011 [doi]
AB  - BACKGROUND: Myasthenia gravis (MG) is an autoimmune disorder of the neuromuscular 
      junction usually caused by antibodies to the nicotinic acetylcholine receptor 
      (AChR) and occasionally to muscle-specific kinase (MuSK). D-penicillamine is a 
      therapeutic agent for several diseases, but can also induce a number of 
      immune-mediated disorders, including MG, as a side-effect. In most patients with 
      D-penicillamine-induced MG, anti-AChR antibodies are detected, but the presence 
      of anti-MuSK antibodies has not been reported previously. CASE: The case reported 
      was a female patient who presented with myasthenic symptoms after D-penicillamine 
      administration for scleroderma. RESULTS: Both anti-AChR and anti-MuSK antibodies 
      were identified in the patient's serum. The anti-MuSK antibodies were of the IgG4 
      subclass, as in idiopathic MG. Both types of antibody gradually disappeared after 
      discontinuation of D-penicillamine. A significant improvement in symptoms was 
      observed and the patient gradually became free of MG symptoms, without requiring 
      any treatment for MG. Another four double-positive (anti-AChR and anti-MuSK 
      antibodies) patients were identified during a retrospective study, but none had 
      been treated with D-penicillamine. CONCLUSION: D-penicillamine can cause 
      anti-AChR and anti-MuSK antibody-positive MG, a rare phenomenon which is reversed 
      after discontinuation of D-penicillamine treatment.
CI  - Copyright (c) 2012 Elsevier B.V. All rights reserved.
FAU - Poulas, Konstantinos
AU  - Poulas K
AD  - Department of Pharmacy, University of Patras, GR 26500, Patras, Greece. 
      kpoulas@upatras.gr
FAU - Koutsouraki, Euphrosyni
AU  - Koutsouraki E
FAU - Kordas, Gregory
AU  - Kordas G
FAU - Kokla, Anna
AU  - Kokla A
FAU - Tzartos, Socrates J
AU  - Tzartos SJ
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Research Support, Non-U.S. Gov't
DEP - 20120609
PL  - Netherlands
TA  - J Neuroimmunol
JT  - Journal of neuroimmunology
JID - 8109498
RN  - 0 (Antirheumatic Agents)
RN  - 0 (Autoantibodies)
RN  - 0 (Autoantigens)
RN  - 0 (Receptors, Cholinergic)
RN  - EC 2.7.10.1 (MUSK protein, human)
RN  - EC 2.7.10.1 (Receptor Protein-Tyrosine Kinases)
RN  - GNN1DV99GX (Penicillamine)
SB  - IM
MH  - Aged
MH  - Antirheumatic Agents/*adverse effects
MH  - Autoantibodies/blood/immunology
MH  - Autoantigens/immunology
MH  - Female
MH  - Humans
MH  - Myasthenia Gravis/*chemically induced/immunology
MH  - Penicillamine/*adverse effects
MH  - Receptor Protein-Tyrosine Kinases/*immunology
MH  - Receptors, Cholinergic/*immunology
MH  - Scleroderma, Systemic/drug therapy/physiopathology
EDAT- 2012/06/12 06:00
MHDA- 2012/10/19 06:00
CRDT- 2012/06/12 06:00
PHST- 2011/11/06 00:00 [received]
PHST- 2012/03/17 00:00 [revised]
PHST- 2012/05/16 00:00 [accepted]
PHST- 2012/06/12 06:00 [entrez]
PHST- 2012/06/12 06:00 [pubmed]
PHST- 2012/10/19 06:00 [medline]
AID - S0165-5728(12)00165-8 [pii]
AID - 10.1016/j.jneuroim.2012.05.011 [doi]
PST - ppublish
SO  - J Neuroimmunol. 2012 Sep 15;250(1-2):94-8. doi: 10.1016/j.jneuroim.2012.05.011. 
      Epub 2012 Jun 9.