Literature DB >> 28028683

Muckle-Wells syndrome in Chinese patients: a single center case series.

Di Wu1, Min Shen2.   

Abstract

Muckle-Wells syndrome (MWS) is a rare autoinflammatory disease. This study aimed to report the clinical features and gene variations of the first case series of MWS patients in Chinese population. Four Han Chinese patients were diagnosed with MWS and followed up at our adult clinic for autoinflammatory diseases. All relevant phenotypes and genotypes were collected. All patients were adult male. The median age of disease onset was 4.5 years, and one patient had adult-onset disease. No positive family history was observed. All patients had a remittent disease course. The duration of fever attacks ranged from 0.5 to 7 days. Skin rashes were present in all patients. The other manifestations included polyarthralgia/arthritis (n = 3), oral ulcers (n = 2), conjunctivitis (n = 2), myalgia (n = 2), headache (n = 2), pharyngitis (n = 1), abdominal pain (n = 1), severe sensorineural hearing loss (n = 1), and chronic meningitis with communicating hydrocephalus (n = 1). None of the patients showed evidence of renal amyloidosis. Each patient carried a heterozygous mutation in an NLRP3 gene, including D29V, V70M, T348M, and Q703K, respectively. D29V and V70M variants were novel mutations in exon 1 of NLRP3. All patients had good response to corticosteroids. Our study suggests that MWS could be identified in Chinese population. Our finding of novel mutations in NLRP3 may expand the diversity of MWS.

Entities:  

Keywords:  Autoinflammatory disease; Cryopyrin-associated periodic syndrome; Muckle-Wells syndrome; NLRP3

Mesh:

Substances:

Year:  2016        PMID: 28028683     DOI: 10.1007/s10067-016-3523-3

Source DB:  PubMed          Journal:  Clin Rheumatol        ISSN: 0770-3198            Impact factor:   2.980


  13 in total

1.  Risk factors for severe Muckle-Wells syndrome.

Authors:  Jasmin B Kümmerle-Deschner; Pascal N Tyrrell; Fabian Reess; Ina Kötter; Peter Lohse; Hermann Girschick; Christian Huemer; Gerd Horneff; Johannes-Peter Haas; Assen Koitschev; Christoph Deuter; Susanne M Benseler
Journal:  Arthritis Rheum       Date:  2010-12

Review 2.  Clinical and genetic characterization of the autoinflammatory diseases diagnosed in an adult reference center.

Authors:  José Hernández-Rodríguez; Estíbaliz Ruíz-Ortiz; Adrià Tomé; Gerard Espinosa; Eva González-Roca; Anna Mensa-Vilaró; Sergio Prieto-González; Georgina Espígol-Frigolé; Josep Mensa; Francesc Cardellach; Josep M Grau; Maria C Cid; Jordi Yagüe; Juan I Aróstegui; Ricard Cervera
Journal:  Autoimmun Rev       Date:  2015-08-21       Impact factor: 9.754

Review 3.  Recommendations for the management of autoinflammatory diseases.

Authors:  Nienke M ter Haar; Marlen Oswald; Jerold Jeyaratnam; Jordi Anton; Karyl S Barron; Paul A Brogan; Luca Cantarini; Caroline Galeotti; Gilles Grateau; Veronique Hentgen; Michael Hofer; Tilmann Kallinich; Isabelle Kone-Paut; Helen J Lachmann; Huri Ozdogan; Seza Ozen; Ricardo Russo; Anna Simon; Yosef Uziel; Carine Wouters; Brian M Feldman; Sebastiaan J Vastert; Nico M Wulffraat; Susanne M Benseler; Joost Frenkel; Marco Gattorno; Jasmin B Kuemmerle-Deschner
Journal:  Ann Rheum Dis       Date:  2015-06-24       Impact factor: 19.103

4.  Long-term clinical course of patients carrying the Q703K mutation in the NLRP3 gene: a case series.

Authors:  Antonio Vitale; Orso Maria Lucherini; Mauro Galeazzi; Bruno Frediani; Luca Cantarini
Journal:  Clin Exp Rheumatol       Date:  2012-12-17       Impact factor: 4.473

5.  Mutations in the autoinflammatory cryopyrin-associated periodic syndrome gene: epidemiological study and lessons from eight years of genetic analysis in France.

Authors:  L Cuisset; I Jeru; B Dumont; A Fabre; E Cochet; J Le Bozec; M Delpech; S Amselem; I Touitou
Journal:  Ann Rheum Dis       Date:  2010-11-24       Impact factor: 19.103

6.  Somatic NLRP3 mosaicism in Muckle-Wells syndrome. A genetic mechanism shared by different phenotypes of cryopyrin-associated periodic syndromes.

Authors:  Kenji Nakagawa; Eva Gonzalez-Roca; Alejandro Souto; Toshinao Kawai; Hiroaki Umebayashi; Josep María Campistol; Jeronima Cañellas; Syuji Takei; Norimoto Kobayashi; Jose Luis Callejas-Rubio; Norberto Ortego-Centeno; Estíbaliz Ruiz-Ortiz; Fina Rius; Jordi Anton; Estibaliz Iglesias; Santiago Jimenez-Treviño; Carmen Vargas; Julian Fernandez-Martin; Inmaculada Calvo; José Hernández-Rodríguez; María Mendez; María Teresa Dordal; Maria Basagaña; Segundo Bujan; Masato Yashiro; Tetsuo Kubota; Ryuji Koike; Naoko Akuta; Kumiko Shimoyama; Naomi Iwata; Megumu K Saito; Osamu Ohara; Naotomo Kambe; Takahiro Yasumi; Kazushi Izawa; Tomoki Kawai; Toshio Heike; Jordi Yagüe; Ryuta Nishikomori; Juan I Aróstegui
Journal:  Ann Rheum Dis       Date:  2013-12-10       Impact factor: 19.103

7.  Hearing loss in Muckle-Wells syndrome.

Authors:  Jasmin B Kuemmerle-Deschner; Assen Koitschev; Katharina Ummenhofer; Sandra Hansmann; Stefan K Plontke; Christiane Koitschev; Ina Koetter; Eva Angermair; Susanne M Benseler
Journal:  Arthritis Rheum       Date:  2013-03

8.  Phenotypic and genotypic characteristics of cryopyrin-associated periodic syndrome: a series of 136 patients from the Eurofever Registry.

Authors:  R Levy; L Gérard; J Kuemmerle-Deschner; H J Lachmann; I Koné-Paut; L Cantarini; P Woo; A Naselli; B Bader-Meunier; A Insalaco; S M Al-Mayouf; S Ozen; M Hofer; J Frenkel; C Modesto; I Nikishina; T Schwarz; S Martino; A Meini; P Quartier; A Martini; N Ruperto; B Neven; M Gattorno
Journal:  Ann Rheum Dis       Date:  2014-07-18       Impact factor: 19.103

9.  Spectrum of clinical features in Muckle-Wells syndrome and response to anakinra.

Authors:  Philip N Hawkins; Helen J Lachmann; Ebun Aganna; Michael F McDermott
Journal:  Arthritis Rheum       Date:  2004-02

10.  Comparison of Tripterygium wilfordii Hook F with methotrexate in the treatment of active rheumatoid arthritis (TRIFRA): a randomised, controlled clinical trial.

Authors:  Qian-Wen Lv; Wen Zhang; Qun Shi; Wen-jie Zheng; Xin Li; Hua Chen; Qing-jun Wu; Wan-lan Jiang; Hong-bin Li; Lu Gong; Wei Wei; Hui Liu; Ai-jing Liu; Hong-tao Jin; Jun-xiang Wang; Xiu-mei Liu; Zhen-bin Li; Bin Liu; Min Shen; Qian Wang; Xiang-ni Wu; Di Liang; Yu-feng Yin; Yun-yun Fei; Jing-mei Su; Li-dan Zhao; Ying Jiang; Jing Li; Fu-lin Tang; Feng-chun Zhang; Peter E Lipsky; Xuan Zhang
Journal:  Ann Rheum Dis       Date:  2014-04-14       Impact factor: 19.103

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  5 in total

1.  Rare mutations in NLRP3 and NLRP12 associated with familial cold autoinflammatory syndrome: two Chinese pedigrees.

Authors:  Shirui Chen; Zhen Li; Xia Hu; Hui Zhang; Weiwei Chen; Qiongqiong Xu; Lili Tang; Huiyao Ge; Qi Zhen; Liang Yong; Yafen Yu; Lu Cao; Ruixue Zhang; Yong Hao; Jihai Shi; Liangdan Sun
Journal:  Clin Rheumatol       Date:  2022-07-19       Impact factor: 3.650

2.  Computational Modeling of NLRP3 Identifies Enhanced ATP Binding and Multimerization in Cryopyrin-Associated Periodic Syndromes.

Authors:  Jenny Mae Samson; Dinoop Ravindran Menon; Prasanna K Vaddi; Nazanin Kalani Williams; Joanne Domenico; Zili Zhai; Donald S Backos; Mayumi Fujita
Journal:  Front Immunol       Date:  2020-11-19       Impact factor: 7.561

Review 3.  Monogenic Adult-Onset Inborn Errors of Immunity.

Authors:  Frederik Staels; Tom Collignon; Albrecht Betrains; Margaux Gerbaux; Mathijs Willemsen; Stephanie Humblet-Baron; Adrian Liston; Steven Vanderschueren; Rik Schrijvers
Journal:  Front Immunol       Date:  2021-11-17       Impact factor: 7.561

4.  Development and Validation of a Targeted Next-Generation Sequencing Gene Panel for Children With Neuroinflammation.

Authors:  Dara McCreary; Ebun Omoyinmi; Ying Hong; Ciara Mulhern; Charalampia Papadopoulou; Marina Casimir; Yael Hacohen; Rodney Nyanhete; Helena Ahlfors; Thomas Cullup; Ming Lim; Kimberly Gilmour; Kshitij Mankad; Evangeline Wassmer; Stefan Berg; Cheryl Hemingway; Paul Brogan; Despina Eleftheriou
Journal:  JAMA Netw Open       Date:  2019-10-02

5.  Ocular manifestations in Chinese adult patients with NLRP3-associated autoinflammatory disease.

Authors:  Tianli Meng; Di Wu; Yi Luo; Na Wu; Mengzhu Zhao; Min Shen; Weihong Yu
Journal:  Sci Rep       Date:  2021-06-07       Impact factor: 4.379

  5 in total

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