[Improvement of sensoneurinal hearing loss in a patient with Muckle-Wells syndrome treated with anakinra]

Klin Padiatr. 2010 Jul;222(4):266-8. doi: 10.1055/s-0029-1239527. Epub 2010 Feb 4.
[Article in German]

Abstract

Muckle Wells syndrome is an autoinflammatory disease in the group of cryopyrin associated periodic syndromes (CAPS). We report the case of an 8 year old girl with MWS who presented with remitting fever, urticaria, remitting coxitis, osteitis, bilateral uveitis anterior, elevated levels of C-reactive protein (CRP) and Serum amyloid A (SAA) and progressive sensoneurinal hearing loss. After starting treatment with anakinra, clinical symptoms dissolved almost completely for about two years now. CRP and SAA levels normalized quickly and sustained and as a consequence the risk of amyloidosis may be minimized. Notable is the complete recovery from sensoneurinal hearing loss merely two months after start of treatment. This brings up questions about pathophysiology of sensoneurinal hearing loss in MWS and emphasizes the benefits of an early diagnosis, as an early start of treatment possibly reduces long-term damage.

Publication types

  • Case Reports

MeSH terms

  • Antirheumatic Agents / therapeutic use*
  • Audiometry, Pure-Tone
  • Auditory Threshold / drug effects*
  • Child
  • Cryopyrin-Associated Periodic Syndromes / diagnosis
  • Cryopyrin-Associated Periodic Syndromes / drug therapy*
  • Cryopyrin-Associated Periodic Syndromes / genetics
  • DNA Mutational Analysis
  • Female
  • Genetic Carrier Screening
  • Hearing Loss, High-Frequency / diagnosis
  • Hearing Loss, High-Frequency / drug therapy*
  • Hearing Loss, High-Frequency / genetics
  • Humans
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use*
  • Long-Term Care

Substances

  • Antirheumatic Agents
  • Interleukin 1 Receptor Antagonist Protein