Literature DB >> 35288824

Multimodal immunotherapy ameliorates myasthenia gravis preceded by thymoma-associated multiorgan autoimmunity.

Hiroo Kasahara1, Kazuki Shimizu1, Kouki Makioka1, Takaaki Sano2, Yoichi Ohtaki3, Yoshio Ikeda4.   

Abstract

Thymoma-associated multiorgan autoimmunity (TAMA) is a rare autoimmune disorder associated with thymoma that causes a pathology similar to graft-versus-host disease (GVHD) targeting the skin, digestive organs, and liver. Herein, we report the case of a 38-year-old male with myasthenia gravis (MG) preceded by TAMA. The patient developed intractable diarrhea 2 years before admission. Subsequently, dysphagia, dysarthria, and left blepharoptosis were observed. The patient was admitted to the hospital because of fever and dyspnea, was positive for anti-AChR antibody, and chest-computed tomography revealed thymoma, which led to the diagnosis of thymoma-related MG. Biopsied specimens from the sigmoid colon revealed apoptotic colonopathy with lymphocyte-rich lamina propria. Immunohistochemical staining revealed that the infiltrating cells were predominantly labeled with anti-CD3-antibody. The patient did not show skin lesions or liver dysfunction. Therefore, TAMA limited to the gastrointestinal tract was diagnosed. Although TAMA typically has a poor prognosis, immediate multimodal immunotherapy for MG was successful, resulting in a good outcome for TAMA of this case. TAMA is caused by the inability of the thymoma to suppress self-reactive T lymphocytes, which subsequently leads to a disease that is clinically indistinguishable from GVHD. Based on the characteristics of this case, limited gastrointestinal tract involvement in TAMA without lesions in other organs may lead to a favorable prognosis. TAMA cases lacking skin lesions may present with nonspecific gastrointestinal or liver disease. If a patient with thymoma-associated MG has gastrointestinal symptoms such as diarrhea, TAMA should be considered, and the diagnosis should be made early by pathological evaluation of gastrointestinal tissues.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

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Year:  2022        PMID: 35288824     DOI: 10.1007/s12026-022-09273-0

Source DB:  PubMed          Journal:  Immunol Res        ISSN: 0257-277X            Impact factor:   2.829


  15 in total

1.  Reactivation of herpes simplex virus and cytomegalovirus in a case of thymoma-associated graft-versus-host disease-like erythroderma.

Authors:  Teruasa Murata; Mizue Yamamoto; Satoshi Kore-eda; Teruhisa Azuma; Kenji Kabashima; Kazumasa Morita
Journal:  Acta Derm Venereol       Date:  2013-11       Impact factor: 4.437

2.  Diminished regulatory T cells in cutaneous lesions of thymoma-associated multi-organ autoimmunity: a newly described paraneoplastic autoimmune disorder with fatal clinical course.

Authors:  T Hanafusa; H Azukizawa; S Kitaba; H Murota; N Umegaki; M Terao; S Sano; T Nakagiri; M Okumura; I Katayama
Journal:  Clin Exp Immunol       Date:  2011-11       Impact factor: 4.330

3.  Thymoma-associated cutaneous graft-versus-host-like disease possibly treated with Narrow-band UVB phototherapy.

Authors:  Minako Nakayama; Munenari Itoh; Sota Kikuchi; Katsumi Tanito; Hidemi Nakagawa
Journal:  Eur J Dermatol       Date:  2016-04-01       Impact factor: 3.328

4.  GVHD-like erythroderma in the clinical course of thymoma-associated myasthenia gravis.

Authors:  Hisashi Nomura; Shigeaki Suzuki; Fumiyo Yasuda-Sekiguchi; Masayuki Amagai; Satoshi Yamada; Jin Nakahara; Norihiro Suzuki; Mitsutomo Kohno; Takeru Funakoshi
Journal:  Eur J Dermatol       Date:  2017-06-01       Impact factor: 3.328

5.  Thymoma-associated multiorgan autoimmunity: a graft-versus-host-like disease.

Authors:  Akhil Wadhera; Emanual Maverakis; Nicholas Mitsiades; Primo N Lara; Maxwell A Fung; Peter J Lynch
Journal:  J Am Acad Dermatol       Date:  2007-04-16       Impact factor: 11.527

6.  Graft-versus-Host disease-like syndrome in malignant thymoma.

Authors:  M H Wang; J M Wong; C Y Wang
Journal:  Scand J Gastroenterol       Date:  2000-06       Impact factor: 2.423

7.  Graft-versus-host-like disease complicating thymoma: lack of AIRE expression as a cause of non-hereditary autoimmunity?

Authors:  G Johan Offerhaus; Marguerite E I Schipper; Audrey J Lazenby; Elizabeth Montgomery; Folkert H M Morsink; Richard J Bende; Alex R Musler; Rene A W van Lier; Carel J M van Noesel
Journal:  Immunol Lett       Date:  2007-09-29       Impact factor: 3.685

8.  Thymoma-associated graft-versus-host disease-like erythroderma.

Authors:  Tomoyuki Nakagiri; Meinoshin Okumura; Masayoshi Inoue; Masato Minami; Eiji Kiyohara; Noriko Umegaki; Toshiaki Nakamura; Yasuhiko Tomita; Ichirou Katayama; Katsuyuki Aozasa
Journal:  J Thorac Oncol       Date:  2007-12       Impact factor: 15.609

9.  Graft-versus-host disease-like erythroderma: a manifestation of thymoma-associated multiorgan autoimmunity.

Authors:  Shay Warren; Kishwer Nehal; Christiane Querfeld; Richard Wong; James Huang; Melissa Pulitzer
Journal:  J Cutan Pathol       Date:  2015-10       Impact factor: 1.587

10.  Graft-versus-host-like colitis and malignant thymoma.

Authors:  Philip W Lowry; John D Myers; Alex Geller; David G Bostwick; Jonathan E Clain
Journal:  Dig Dis Sci       Date:  2002-09       Impact factor: 3.487

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