Literature DB >> 25998611

Selective response to rituximab in a young child with MuSK-associated myasthenia gravis.

Raghav Govindarajan1, Stanley J Iyadurai2, Anne Connolly3, Craig Zaidman3.   

Abstract

Neuromuscular junction disorders in children are either genetic, such as congenital myasthenic syndrome, or autoimmune with circulating antibodies most commonly against acetylcholine receptors. There is limited experience recognizing and treating children with myasthenia associated with muscle-specific tyrosine kinase antibodies. We report a seven-year-old child with intermittent esotropia since age 3 months, and two years of progressive and severe diplopia, dysarthria, dysphagia, and facial weakness. Acetylcholine receptor antibodies and genetic testing for congenital myasthenic syndrome were negative. Muscle specific tyrosine kinase antibodies were significantly elevated. Ophthalmoplegia and bulbar weakness were refractory to treatment with acetylcholinesterase inhibitors, corticosteroids and IVIg but completely resolved following treatment with rituximab. Her neurologic examination remained normal at the most recent follow-up, 15 months after initiation of rituximab. Children with MuSK myasthenia, like adults, can respond to rituximab despite long standing disease and failure to improve on other immunosuppressant medications.
Copyright © 2015 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Diplopia; Dysarthria; Muscle specific tyrosine kinase; Myasthenia gravis; Rituximab

Mesh:

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Year:  2015        PMID: 25998611     DOI: 10.1016/j.nmd.2015.03.014

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  2 in total

Review 1.  Mechanisms underlying B cell immune dysregulation and autoantibody production in MuSK myasthenia gravis.

Authors:  Panos Stathopoulos; Aditya Kumar; Jason A Vander Heiden; Elba Pascual-Goñi; Richard J Nowak; Kevin C O'Connor
Journal:  Ann N Y Acad Sci       Date:  2018-01       Impact factor: 5.691

Review 2.  Muscle-Specific Kinase Myasthenia Gravis.

Authors:  Lucia S Borges; David P Richman
Journal:  Front Immunol       Date:  2020-05-08       Impact factor: 7.561

  2 in total

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