Muckle-Wells syndrome: a treatable cause of congenital sensorineural hearing loss

被引:0
作者
Stew, B. T. [1 ]
Fishpool, S. J. C. [1 ]
Owens, D. [2 ]
Quine, S. [2 ]
机构
[1] Singleton Hosp, Dept ENT Surg, Swansea SA2 8QA, W Glam, Wales
[2] Univ Wales Hosp, Dept ENT Surg, Cardiff CF4 4XW, S Glam, Wales
来源
B-ENT | 2013年 / 9卷 / 02期
关键词
Muckle; Wells; congenital hearing loss; sensorineural; anakinra; ANAKINRA; DEAFNESS; PATIENT;
D O I
暂无
中图分类号
R76 [耳鼻咽喉科学];
学科分类号
100213 ;
摘要
Muckle-Wells syndrome (MWS) is a rare autosomal dominant condition with variable expression. It is a subset of auto-inflammatory diseases characterised by recurrent inflammatory crises and is associated with chronic recurrent urticaria, sensorineural deafness, periodic arthritis and secondary amyloidosis. The diagnosis of MWS is a clinical one with sufferers classically presenting in childhood with a moderate fever and non-pruiginous urticaria. We describe a case of a six-year-old girl who was successfully diagnosed and treated with Anakinra. Muckle and Wells originally described this syndrome in 1962; however, only recently was it discovered to be genetically linked to chromosome 1q44 and subsequently to missense mutations in the CIAS1/NALP3/PYPAF1 gene. Since then, treatment has evolved and it remains one of few treatable causes of congenital profound sensorineural hearing loss.
引用
收藏
页码:161 / 163
页数:3
相关论文
共 9 条
  • [1] Spectrum of clinical features in Muckle-Wells syndrome and response to anakinra
    Hawkins, PN
    Lachmann, HJ
    Aganna, E
    McDermott, MF
    [J]. ARTHRITIS AND RHEUMATISM, 2004, 50 (02): : 607 - 612
  • [2] Interleukin-1-receptor antagonist in the Muckle-Wells syndrome
    Hawkins, PN
    Lachmann, HJ
    McDermott, MF
    [J]. NEW ENGLAND JOURNAL OF MEDICINE, 2003, 348 (25) : 2583 - 2584
  • [3] Clinical aspects of hereditary hearing loss
    Kochhar, Amit
    Hildebrand, Michael S.
    Smith, Richard J. H.
    [J]. GENETICS IN MEDICINE, 2007, 9 (07) : 393 - 408
  • [4] Efficacy and Safety of Anakinra Therapy in Pediatric and Adult Patients With the Autoinflammatory Muckle-Wells Syndrome
    Kuemmerle-Deschner, Jasmin B.
    Tyrrell, Pascal N.
    Koetter, Ina
    Wittkowski, Helmut
    Bialkowski, Anja
    Tzaribachev, Nicolai
    Lohse, Peter
    Koitchev, Assen
    Deuter, Christoph
    Foell, Dirk
    Benseler, Susanne M.
    [J]. ARTHRITIS AND RHEUMATISM, 2011, 63 (03): : 840 - 849
  • [5] Recovery from deafness in a patient with Muckle-Wells syndrome treated with anakinra
    Mirault, Tristan
    Launay, David
    Cuisset, Laurence
    Hachulla, Eric
    Lambert, Marc
    Queyrel, Viviane
    Quemeneur, Thomas
    Morell-Dubois, Sandrine
    Hatron, Pierre-Yves
    [J]. ARTHRITIS AND RHEUMATISM, 2006, 54 (05): : 1697 - 1700
  • [6] MUCKLE TJ, 1962, Q J MED, V31, P235
  • [7] Hearing improvement in a patient with variant Muckle-Wells syndrome in response to interleukin 1 receptor antagonism
    Rynne, M
    Maclean, C
    Bybee, A
    McDermott, MF
    Emery, P
    [J]. ANNALS OF THE RHEUMATIC DISEASES, 2006, 65 (04) : 533 - 534
  • [8] Savastano M, 2010, B-ENT, V6, P183
  • [9] Anakinra improves sensory deafness in a Japanese patient with Muckle-Wells syndrome, possibly by inhibiting the cryopyrin inflammasome
    Yamazaki, Takashi
    Masumoto, Junya
    Agematsu, Kazunaga
    Sawai, Nobukuni
    Kobayashi, Shinji
    Shigemura, Tomonari
    Yasui, Kozo
    Koike, Kenichi
    [J]. ARTHRITIS AND RHEUMATISM, 2008, 58 (03): : 864 - 868