Literature DB >> 28364296

Juvenile-onset myasthenia gravis: autoantibody status, clinical characteristics and genetic polymorphisms.

Yu Hong1, Geir Olve Skeie1,2, Paraskevi Zisimopoulou3, Katerina Karagiorgou3,4, Socrates J Tzartos3,4, Xiang Gao5, Yao-Xian Yue6, Fredrik Romi1,2, Xu Zhang5, Hai-Feng Li7, Nils Erik Gilhus8,9.   

Abstract

Myasthenia gravis (MG) is an autoimmune disorder mediated by antibodies against proteins at the neuromuscular junction. Juvenile-onset MG (JMG) has been reported to have special characteristics. It is still unclear whether there are any pathogenic and genetic differences between juvenile and adult MG. In this study, we evaluated the clinical characteristics, autoantibody status (antibodies against AChR, MuSK, LRP4, titin and RyR) and genetic susceptibility (CHRNA1, CTLA4 and AIRE) in 114 Chinese JMG patients, and compared with 207 young adult MG patients (onset age 18-40 years). JMG patients were classified into two subgroups: the very early onset group (<8 years) and puberty onset group (8-18 years). The very early onset MG patients had a higher proportion of ocular MG and thymus hyperplasia, compared with puberty onset MG and young adult MG (P < 0.05). AChR antibodies were found in majority of JMG patients and were associated with more severe disease (P < 0.05), while other antibodies were rare in JMG. Moreover, the very early onset MG had a more prominent genetic predisposition than puberty and adult MG, affecting the susceptible genes CHRNA1 and CTLA4. JMG has the same pathogenic background as adult MG, but has typical clinical features and a prominent genetic predisposition in very early onset patients (<8 years). Specific therapeutic considerations are needed.

Entities:  

Keywords:  Acetylcholine receptor; Cholinergic receptor nicotinic alpha 1 (CHRNA1); Cytotoxic T-lymphocyte-associated protein 4 (CTLA-4); Gene polymorphisms; Juvenile onset myasthenia gravis; Muscle specific kinase

Mesh:

Substances:

Year:  2017        PMID: 28364296     DOI: 10.1007/s00415-017-8478-z

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  37 in total

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Review 2.  Myasthenia gravis - autoantibody characteristics and their implications for therapy.

Authors:  Nils Erik Gilhus; Geir Olve Skeie; Fredrik Romi; Konstantinos Lazaridis; Paraskevi Zisimopoulou; Socrates Tzartos
Journal:  Nat Rev Neurol       Date:  2016-04-22       Impact factor: 42.937

Review 3.  Myasthenia gravis: a comprehensive review of immune dysregulation and etiological mechanisms.

Authors:  Sonia Berrih-Aknin; Rozen Le Panse
Journal:  J Autoimmun       Date:  2014-01-03       Impact factor: 7.094

4.  [Antigens of histocompatibility in children with myasthenia gravis].

Authors:  V Ia Neretin; L D Serova; B V Agafonov; V G Tsuman; B M Gekht; O P Sidorova; A E Nalivkin
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5.  MuSK autoantibodies in myasthenia gravis detected by cell based assay--A multinational study.

Authors:  A I Tsonis; P Zisimopoulou; K Lazaridis; J Tzartos; E Matsigkou; V Zouvelou; R Mantegazza; C Antozzi; F Andreetta; A Evoli; F Deymeer; G Saruhan-Direskeneli; H Durmus; T Brenner; A Vaknin; S Berrih-Aknin; A Behin; T Sharshar; M De Baets; M Losen; P Martinez-Martinez; K A Kleopa; E Zamba-Papanicolaou; T Kyriakides; A Kostera-Pruszczyk; P Szczudlik; B Szyluk; D Lavrnic; I Basta; S Peric; C Tallaksen; A Maniaol; C Casasnovas Pons; J Pitha; M Jakubíkova; F Hanisch; S J Tzartos
Journal:  J Neuroimmunol       Date:  2015-04-27       Impact factor: 3.478

6.  A cohort study on myasthenia gravis patients in China.

Authors:  Wei Wang; Yu-Ping Chen; Zhong-Kui Wang; Dong-Ning Wei; Ling Yin
Journal:  Neurol Sci       Date:  2013-02-20       Impact factor: 3.307

7.  Expression of interleukin-22 in myasthenia gravis.

Authors:  S Zheng; C Dou; N Xin; J Wang; J Wang; P Li; L Fu; X Shen; G Cui; R Dong; J Lu; Y Zhang
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8.  Autoantibody profile and clinical characteristics in a cohort of Chinese adult myasthenia gravis patients.

Authors:  Yu Hong; Hai-Feng Li; Geir Olve Skeie; Fredrik Romi; Hong-Jun Hao; Xu Zhang; Xiang Gao; Jone Furlund Owe; Nils Erik Gilhus
Journal:  J Neuroimmunol       Date:  2016-07-02       Impact factor: 3.478

9.  HLA-DRB1*14 and DQB1*05 are associated with Japanese anti-MuSK antibody-positive myasthenia gravis patients.

Authors:  Tetsuya Kanai; Akiyuki Uzawa; Naoki Kawaguchi; Tateo Sakamaki; Yasumasa Yoshiyama; Keiichi Himuro; Fumiko Oda; Satoshi Kuwabara
Journal:  J Neurol Sci       Date:  2016-02-16       Impact factor: 3.181

10.  Human muscle acetylcholine receptor alpha-subunit gene (CHRNA1) association with autoimmune myasthenia gravis in black, mixed-ancestry and Caucasian subjects.

Authors:  J M Heckmann; K E Morrison; B Emeryk-Szajewska; H Strugalska; J Bergoffen; N Willcox; J Newsom-Davis
Journal:  J Autoimmun       Date:  1996-04       Impact factor: 7.094

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  6 in total

1.  User Involvement in Myasthenia Gravis Research.

Authors:  Nils Erik Gilhus; Sandra Iren Barkås Hovland
Journal:  Front Neurol       Date:  2022-06-02       Impact factor: 4.086

2.  Retraction Note to: Juvenile-onset myasthenia gravis: autoantibody status, clinical characteristics and genetic polymorphisms.

Authors:  Yu Hong; Geir Olve Skeie; Paraskevi Zisimopoulou; Katerina Karagiorgou; Socrates J Tzartos; Xiang Gao; Yao-Xian Yue; Fredrik Romi; Xu Zhang; Hai-Feng Li; Nils Erik Gilhus
Journal:  J Neurol       Date:  2019-06       Impact factor: 4.849

Review 3.  Nuclear Factor-kappaB in Autoimmunity: Man and Mouse.

Authors:  Bahar Miraghazadeh; Matthew C Cook
Journal:  Front Immunol       Date:  2018-04-09       Impact factor: 7.561

Review 4.  Myasthenia Gravis Can Have Consequences for Pregnancy and the Developing Child.

Authors:  Nils Erik Gilhus
Journal:  Front Neurol       Date:  2020-06-12       Impact factor: 4.003

5.  Long-Term Improvement in a Chinese Cohort of Glucocorticoid-Resistant Childhood-Onset Myasthenia Gravis Patients Treated With Tacrolimus.

Authors:  Zhuajin Bi; Yayun Cao; Jing Lin; Qing Zhang; Chenchen Liu; Mengcui Gui; Bitao Bu
Journal:  Front Neurol       Date:  2022-02-08       Impact factor: 4.003

6.  Immune checkpoint inhibitors in the onset of myasthenia gravis with hyperCKemia.

Authors:  Koutaro Takamatsu; Shunya Nakane; Shigeaki Suzuki; Takayuki Kosaka; Satoshi Fukushima; Toshihiro Kimura; Azusa Miyashita; Akihiro Mukaino; Shiori Yamakawa; Keisuke Watanabe; Masatoshi Jinnin; Yoshihiro Komohara; Hironobu Ihn; Yukio Ando
Journal:  Ann Clin Transl Neurol       Date:  2018-09-23       Impact factor: 4.511

  6 in total

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