Abstract |
Hyperimmunoglobulin D and periodic fever syndrome (HIDS) is a rare, autoinflammatory condition caused by mutations in the mevalonate kinase gene. There is no standard treatment for HIDS, and randomized controlled trials are lacking. Corticosteroids, colchicine, nonsteroidal anti-inflammatory drugs, statins, and cyclosporine are of limited efficacy in controlling this condition. Recent case reports suggest that most patients respond to etanercept or anakinra. Interleukin 6 blockade in HIDS has not been described. We report the case of a 13-year-old girl with HIDS, who failed to respond to colchicine, corticosteroids, etanercept, and anakinra but was successfully treated with the anti-IL-6 monoclonal antibody, tocilizumab.
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Authors | Hiba M Shendi, Lisa A Devlin, John David Edgar |
Journal | Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases
(J Clin Rheumatol)
Vol. 20
Issue 2
Pg. 103-5
(Mar 2014)
ISSN: 1536-7355 [Electronic] United States |
PMID | 24561416
(Publication Type: Case Reports, Journal Article)
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Chemical References |
- Antibodies, Monoclonal, Humanized
- Immunoglobulin G
- Interleukin 1 Receptor Antagonist Protein
- Interleukin-6
- Receptors, Tumor Necrosis Factor
- tocilizumab
- Etanercept
- Colchicine
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Topics |
- Adolescent
- Antibodies, Monoclonal, Humanized
(immunology, therapeutic use)
- Colchicine
(therapeutic use)
- Etanercept
- Female
- Humans
- Immunoglobulin G
(therapeutic use)
- Interleukin 1 Receptor Antagonist Protein
(therapeutic use)
- Interleukin-6
(antagonists & inhibitors, immunology)
- Mevalonate Kinase Deficiency
(drug therapy)
- Receptors, Tumor Necrosis Factor
(therapeutic use)
- Treatment Failure
- Treatment Outcome
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