Literature DB >> 25283823

[A case of myasthenia gravis with invasive thymoma associated with diffuse panbronchiolitis, alopecia, dysgeusia, cholangitis and myositis].

Risa Maekawa1, Hideki Shibuya, Takuto Hideyama, Yasushi Shiio.   

Abstract

A 43-year-old man was admitted to our hospital because of diplopia, ptosis, and dysphagia that had begun three years previously. He was diagnosed with myasthenia gravis (MG) and invasive thymoma and treated with corticosteroid, thymectomy, and radiation therapy. Ten years after the thymectomy, computed tomography (CT) showed metastasis of the thymoma in the left lower lobe of the lung. Two years after this recurrence, when the patient was 55, respiratory symptoms such as wheezing, persistent cough, and dyspnea appeared. Chronic sinusitis, diffuse centrilobular opacities on CT, and positivity for HLA-B54 led to a diagnosis of diffuse panbronchiolitis (DPB). Despite treatment with clarithromycin, the respiratory symptoms worsened. The patient developed alopecia and body hair loss at the age of 56 followed by dysgeusia, cholangitis, and myositis with positivity for anti-Kv1.4 antibodies. Although treatment with an increased dose of corticosteroid improved hair loss, dysgeusia, cholangitis, and myositis, he died of progression of DPB and serious respiratory infection at the age of 58. In this case, various autoimmune disorders occurred together with MG as complications of thymoma. Although alopecia, dysgeusia, and myositis are already known as complications of MG associated with thymoma, cholangitis is not well-recognized since there have been few reports suggesting a causal relationship between cholangitis and thymoma. Furthermore, DPB caused by immunodeficiency and respiratory tract hypersensitivity associated with thymoma and HLA-B54, respectively, is the distinctive feature of our case. Neurologists should be aware that various organs can be damaged directly and indirectly by abnormal T cells from thymoma in patients with MG.

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Year:  2014        PMID: 25283823     DOI: 10.5692/clinicalneurol.54.703

Source DB:  PubMed          Journal:  Rinsho Shinkeigaku        ISSN: 0009-918X


  3 in total

1.  A 47-Year-Old Japanese Woman with Symptoms of Increased Salty and Reduced Sweet Taste Perception Preceding a Diagnosis of Thymoma-Associated Myasthenia Gravis.

Authors:  Motoya Kimura; Hiroaki Nakagawa; Jun-Ichi Niwa; Yoshinori Wakita; Yasushi Miyata; Masato Maekawa; Manabu Doyu
Journal:  Am J Case Rep       Date:  2022-05-02

2.  Inflammatory myopathy with myasthenia gravis: Thymoma association and polymyositis pathology.

Authors:  Naohiro Uchio; Kenichiro Taira; Chiseko Ikenaga; Masato Kadoya; Atsushi Unuma; Kenji Yoshida; Setsu Nakatani-Enomoto; Yuki Hatanaka; Yasuhisa Sakurai; Yasushi Shiio; Kenichi Kaida; Akatsuki Kubota; Tatsushi Toda; Jun Shimizu
Journal:  Neurol Neuroimmunol Neuroinflamm       Date:  2018-12-24

3.  Thymic neoplasms patients complicated with bronchiectasis: Case series in a Chinese hospital and literature review.

Authors:  Yongjian Liu; Yan Xu; Xinlun Tian; Hui Huang; Xiaomeng Hou; Minjiang Chen; Wei Zhong; Jing Zhao; Jinming Gao; Jinglan Wang; Juhong Shi; Mengzhao Wang
Journal:  Thorac Cancer       Date:  2019-02-13       Impact factor: 3.500

  3 in total

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