Arthritis & Rheumatism, Volume 62,
November 2010 Abstract Supplement

Abstracts of the American College of
Rheumatology/Association of Rheumatology Health Professionals
Annual Scientific Meeting
Atlanta, Georgia November 6-11, 2010.


Use of TNF Antagonists in the Treatment of Chronic Non-Bacterial Osteomyelitis (CNO).

Stern2,  Sara, Marzan2,  Katherine, Borzutzky1,  Arturo, Steinberg3,  Evan, Reiff2,  Andreas

Boston Children's Hospital, Boston, MA
Childrens Hospital Los Angeles, Los Angeles, CA
Kaiser-Permanente Sunset Hospital, Los Angeles, CA

Purpose:

CNO is a chronic autoimmune inflammatory osteopathy syndrome that is culture negative and has no demonstrable organism on histopathology. Anti-inflammatory medications are typically used to prevent pathologic fractures, pain, and disease relapse. The objective of this study was to describe the use of TNFa antagonists in the treatment of CNO in 8 patients with treatment refractory disease.

Methods:

We retrospectively analyzed 23 patients with CNO seen from 1985–2009 at Childrens Hospital Los Angeles (18) and Kaiser-Permanente Sunset Hospital Los Angeles, CA, USA (5). CNO disease activity was evaluated by clinical, laboratory and radiographic parameters. Response to treatment was assessed by laboratory values (CRP, ESR) and a global response score, which also included clinical and radiographic parameters. A score of 0 indicated lack of response, 1 indicated partial response, and 2 indicated full response. Descriptive statistics were used for analysis.

Results:

23 patients (14 F: 9M) with a mean age of 9.6 ± 2.9 were followed for a median of 1.83 (0.1–13) years. Patients had a mean of 3.3 ± 2.5 lesions which were most commonly seen in the pelvic bone (9), tibia (8), clavicle (6), calcaneus (5), femur (5), humerus (5), vertebrae (5), and mandible (4). Eight patients (34.7%) had unifocal disease. The most common presenting symptoms included bone pain (90.9%), soft tissue swelling (45.5%), and fever (29.4%). Extra osseous manifestations were observed in 7 patients including enthesitis (2), myositis/fasciitis (4), anterior uveitis (1), oral ulcers (1), discoid rash (1) and Crohn's disease (1). Pathologic fractures were seen in 11 (47.8%) patients. All but one patient received NSAIDS, which was sufficient to control disease activity in 7 (31.8%) of patients. Eight patients were treated with methotrexate of which 7 failed this treatment and 5 were subsequently switched to TNFa antagonists of whom 3 improved. The overall failure rate in the anti TNFa group was 50%.

Conclusions:

CNO is a chronic autoimmune inflammatory syndrome with a high risk for pathologic fractures and systemic morbidity. Treatment of these patients remains challenging since they tend to respond poorly to traditional NSAID and DMARD therapy. TNFa antagonists offer a viable treatment option and appeared to be the most beneficial in our study.

  Response
  GlobalClinicalRadiologic ImagesLaboratory
NSAIDs n=22Lack of Response4 (17.4%)3 (13.0%)7 (48.7%)6 (30.0%)
 Partial Response12 (52.2%)11 (47.8%)6 (40.0%)6 (30.0%)
 Full Response7 (30.4%)9 (39.1%)2 (13.3%)8 (40.0%)
Methotrexate n=8Lack of Response2 (25.0%)2 (25.0%)2 (33.3%)5 (62.5%)
 Partial Response5 (62.5%)5 (62.5%)3 (50.0%)2 (25.0%)
 Full Response1 (12.5%)1 (12.5%)1 (16.7%)1 (12.5%)
TNFa Antagonists n=8Lack of Response0 (0%)0 (0%)0 (0%)2 (28.6%)
 Partial Response4 (50.0%)5 (62.5%)3 (60.0%)4 (67.1%)
 Full Response4 (50.0%)3 (37.5%)2 (40.0%)1 (14.3%)

To cite this abstract, please use the following information:
Stern, Sara, Marzan, Katherine, Borzutzky, Arturo, Steinberg, Evan, Reiff, Andreas; Use of TNF Antagonists in the Treatment of Chronic Non-Bacterial Osteomyelitis (CNO). [abstract]. Arthritis Rheum 2010;62 Suppl 10 :241
DOI: 10.1002/art.28010

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