Literature DB >> 29850899

Giant Cell Polymyositis and Myocarditis in a Patient With Thymoma and Myasthenia Gravis: A Postviral Autoimmune Process?

David S Priemer1, Darrell D Davidson1, Patrick J Loehrer2, Sunil S Badve1.   

Abstract

Thymomas are associated with autoantibody formation. The most common are anti-acetylcholine receptor antibodies, which correspond to myasthenia gravis (MG). Other autoantibodies, such as antistriational antibodies, can occur, but their relation to clinical syndromes is frequently uncertain. The etiology of antistriational antibodies is also poorly understood. In this case, a 61-year-old man with a history of thymoma was admitted with respiratory failure. The patient was positive for anti-acetylcholine receptor antibodies and antistriational antibodies. He developed cardiogenic shock and died within 2 days despite aggressive therapy. Laboratory studies revealed elevated cardiac enzymes and marked IgG elevation against Coxsackie A virus serotypes 9 and 24. Subclinical IgG elevations against additional Coxsackie A and Coxsackie B virus serotypes were also noted. Autopsy revealed lymphohistiocytic infiltrates with multinucleated giant cells in the myocardium and skeletal muscles, including the diaphragm. Giant cell polymyositis and myocarditis is a rare, lethal complication in patients with thymoma and MG. The pathogenesis is uncertain. An autoimmune process, possibly elicited by antistriational antibodies, has been suggested. The coexistence of antistriational antibodies and Coxsackie viral serologies has not been reported. This case may suggest that giant cell polymyositis and myocarditis in patients with thymoma and MG is a postviral autoimmune process.

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Year:  2018        PMID: 29850899     DOI: 10.1093/jnen/nly041

Source DB:  PubMed          Journal:  J Neuropathol Exp Neurol        ISSN: 0022-3069            Impact factor:   3.685


  3 in total

Review 1.  Giant cell myositis associated with concurrent myasthenia gravis: a case-based review of the literature.

Authors:  Frank A Scangarello; Luisa Angel-Buitrago; Melanie Lang-Orsini; Alexander Geevarghese; Knarik Arkun; Oscar Soto; Mithila Vullaganti; Robert Kalish
Journal:  Clin Rheumatol       Date:  2021-02-25       Impact factor: 2.980

2.  Immune checkpoint inhibitors for treatment of thymic epithelial tumors: how to maximize benefit and optimize risk?

Authors:  Chen Zhao; Arun Rajan
Journal:  Mediastinum       Date:  2019-09-09

3.  Acquired myasthenia gravis with concurrent polymyositis and myocarditis secondary to a thymoma in a dog.

Authors:  Raffaella Perillo; Marika Menchetti; Pasquale A Giannuzzi; Angela Marchiori; Marco Rondena; Stefania Gasparini
Journal:  Open Vet J       Date:  2021-08-22
  3 in total

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