» Articles » PMID: 21769488

Adult-onset Still's Disease and Chronic Recurrent Multifocal Osteomyelitis: a Hitherto Undescribed Manifestation of Autoinflammation

Overview
Journal Rheumatol Int
Specialty Rheumatology
Date 2011 Jul 20
PMID 21769488
Citations 17
Authors
Affiliations
Soon will be listed here.
Abstract

Still's disease and chronic recurrent multifocal osteomyelitis (CRMO) are febrile rheumatic diseases of unknown etiology, which predominantly affect children but can also have their initial manifestation in adults. Both can present as intermittent, relapsing episodes and are considered potential candidates within the expanding spectrum of autoinflammatory disorders, although no genetic abnormalities have been described for either of them. Here, we describe a man with an initial manifestation of abacterial multifocal osteitis at the age of 41. During a relapsing-remitting course of his illness, he increasingly developed symptoms of adult-onset Still's disease (AOSD), and the diagnosis was established according to the Yamaguchi criteria. When treated with anakinra, not only the acute symptoms disappeared promptly, but also the osteitis went into complete remission. This is to our knowledge the first description of a simultaneous occurrence of these two manifestations of autoinflammation in adulthood.

Citing Articles

Sterile osteomyelitis: a cardinal sign of autoinflammation.

Borges T, Santos J, Silva S Reumatologia. 2025; 62(6):475-488.

PMID: 39866303 PMC: 11758105. DOI: 10.5114/reum/196595.


Juvenile idiopathic arthritis with systemic onset with inflammatory bone lesions: two case reports of patients successfully treated with canakinumab and experience gained from literature.

Alexeeva E, Dvoryakovskaya T, Tsulukiya I, Kondrateva N, Solomatina N, Kondratiev G Front Pediatr. 2023; 11:1163483.

PMID: 37325364 PMC: 10266530. DOI: 10.3389/fped.2023.1163483.


New insights on multigenic autoinflammatory diseases.

Efthimiou P, Petryna O, Nakasato P, Kontzias A Ther Adv Musculoskelet Dis. 2022; 14:1759720X221117880.

PMID: 36081748 PMC: 9445512. DOI: 10.1177/1759720X221117880.


New Insights in the Treatment of SAPHO Syndrome and Medication Recommendations.

Cheng W, Li F, Tian J, Xie X, Chen J, Peng X J Inflamm Res. 2022; 15:2365-2380.

PMID: 35444448 PMC: 9013916. DOI: 10.2147/JIR.S353539.


Chronic Multifocal Osteomyelitis - Is Mycobacterium avium Complex Really the Culprit? A Case Report in an Adult Female.

Johnson J, Gupton M, Finn J, Deivaraju C J Orthop Case Rep. 2022; 11(9):86-89.

PMID: 35415176 PMC: 8930344. DOI: 10.13107/jocr.2021.v11.i09.2428.


References
1.
Fautrel B . Adult-onset Still disease. Best Pract Res Clin Rheumatol. 2008; 22(5):773-92. DOI: 10.1016/j.berh.2008.08.006. View

2.
Ferguson P, El-Shanti H . Autoinflammatory bone disorders. Curr Opin Rheumatol. 2007; 19(5):492-8. DOI: 10.1097/BOR.0b013e32825f5492. View

3.
Klein A, Horneff G . [Improvement of sensoneurinal hearing loss in a patient with Muckle-Wells syndrome treated with anakinra]. Klin Padiatr. 2010; 222(4):266-8. DOI: 10.1055/s-0029-1239527. View

4.
Aksentijevich I, Masters S, Ferguson P, Dancey P, Frenkel J, van Royen-Kerkhoff A . An autoinflammatory disease with deficiency of the interleukin-1-receptor antagonist. N Engl J Med. 2009; 360(23):2426-37. PMC: 2876877. DOI: 10.1056/NEJMoa0807865. View

5.
de Koning H, Bodar E, van der Meer J, Simon A . Schnitzler syndrome: beyond the case reports: review and follow-up of 94 patients with an emphasis on prognosis and treatment. Semin Arthritis Rheum. 2007; 37(3):137-48. DOI: 10.1016/j.semarthrit.2007.04.001. View