Efficacy of Antibiotic Therapy for SAPHO Syndrome is Lost after Its Discontinuation: An Interventional Study

Gunter Assmann; Olaf Kueck; Timm Kirchhoff; Herbert Rosenthal; Jan Voswinkel; Michael Pfreundschuh; Henning Zeidler; Annette D Wagner

Disclosures

Arthritis Res Ther. 2009;11(5):R140 

In This Article

Abstract and Introduction

Abstract

Introduction The acronym SAPHO was introduced in 1987 to unify the various descriptions of a seronegative arthritis associated with skin manifestations and to show synovitis, acne, pustulosis, hyperostosis, and osteitis with and without sterile multifocal osteomyelitis. The etiology of SAPHO syndrome is unknown, but an association with infection by semipathogenic bacteria like Propionibacterium acnes has been suggested. We conducted an interventional study of SAPHO patients receiving antibiotics.
Methods Thirty-seven patients met the clinical criteria of SAPHO syndrome, 21 of them underwent a needle biopsy of the osteitis lesion, and 14 of them showed positive bacteriological cultures for P. acnes. Thirty patients (14 bacteriological positive and 16 without biopsy) were treated with antibiotics for 16 weeks. The activity of skin disease and osteitis were assessed by a physician using a scoring model (from 0 to 6). In addition, patients completed a Health Assessment Score (HAS, from 0 to 6). The erythrocyte sedimentation rate was determined and a MRI (of the osteitis lesion, radiologic activity score from 0 to 2) was performed in week 1 (W1), week 16 (W16), and week 28 (W28, 12 weeks after antibiotics).
Results Twenty-seven patients continued the medication (azithromycin, n = 25, 500 mg twice a week; clindamycin, n = 1, 300 mg daily; or doxycycline, n = 1, 100 mg daily) for 16 weeks. After W16 the scores for MRI (1.5 to 1.1, P = 0.01), skin activity (3.2 to 1.2, P = 0.01), osteitis activity (4.0 to 2.1, P = 0.02), and HAS (3.3 to 2.1, P = 0.01) decreased significantly. However, this was followed by increasing values for MRI scores (1.2 to 1.4, P = 0.08), skin activity (1.2 to 1.7, P = 0.11), osteitis activity (1.9 to 2.7, P = 0.01), and HAS (2.2 to 3.3, P = 0.02) from W16 to W28. The comparison of the scores in W1 and W28 in these 12 patients showed no significant differences.
Conclusions For the period of application, the antibiotic therapy seems to have controlled the disease. After antibiotic discontinuation, however, disease relapse was observed. SAPHO syndrome thus groups with other chronic inflammatory arthropathies with a need for permanent therapy.

Introduction

In 1987 Chamot and colleagues coined the acronym SAPHO for synovitis, acne, pustulosis, hyperostosis, and osteitis, which replaced various previously suggested descriptions of osteoarticular disease associated with skin manifestations.[1,2] The clinical feature of chronic recurrent multiple osteomyelitis (CRMO) with its typical presentation in the pediatric population justifies the inclusion of CRMO into the same nosologic group as the SAPHO syndrome according to several authors.[3,4]

The etiology of these diseases is still unknown. An association with infection by semipathogenic bacteria such as Propionibacterium acnes has been suggested, but the role of these bacteria is discussed controversially.[5,6] Furthermore, a part of coagulase-negative Staphyloccocus aureus as well as Haemophilus parainfluenzae and Actinomyces were reported to be associated with SAPHO syndrome.[7,8] Family-based observations and investigations of genetic variations gave rise to the hypothesis that genetic factors contribute to the development and course of the disease.[9,10] Moreover, SAPHO syndrome shows a clear overlap with several inflammatory rheumatic diseases such as ankylosing spondylitis, psoriatic arthritis, enteropathic arthritis, reactive arthritis, and undifferentiated spondyloarthritis. In 13 to 52% of SAPHO cases, radiologic findings show sacroiliitis - as in typical ankylosing spondylitis.[11] The genetic marker HLA-B27, however, is not clearly associated with SAPHO syndrome.[12,13] Clinical symptoms of psoriatic arthritis are comparable with the features of SAPHO syndrome. In some cases, psoriasis vulgaris has developed after initial typical skin changes in patients with SAPHO syndrome. The skin manifestation of acne vulgaris is not typical of psoriatic arthritis, however, and the psoriatic-typical nail dystrophy has not been reported in SAPHO patients.

Although the classification of SAPHO syndrome exists as a distinct disease entity, the overlap and similarities with other rheumatic diseases formed the basis for trials investigating antirheumatic drugs that are the accepted standard for the treatment of psoriatic arthritis and other spondyloarthritides. Studies have been published with small numbers of patients treated with nonsteroidal anti-inflammatory drugs,[14] steroids[15,16] and immunosuppressive agents that showed only partial efficacy. Investigations of methotrexate and azathioprine yielded no convincing results.[17,18] Several reports presenting promising results obtained with bisphosphonates[19–21] or biologicals like TNFα-blockers,[22,23] however, have recently been published. With regard to the possible link to an infectious etiology of SAPHO syndrome, several studies with small numbers of patients treated with antibiotics reported contradictory results.[24,25] According to these studies, the antibiotic agent of azithromycin was suggested as the most promising agent for treatment of patients with SAPHO syndrome. We have therefore conducted a prospective interventional study to evaluate the efficacy of antibiotics in patients with SAPHO syndrome.

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