Journal List > J Rheum Dis > v.22(5) > 1064217

Shim, Park, Kim, Jung, Baek, Lim, Joo, and Kwon: A Case Report of SAPHO Syndrome Treated with Oral Alendronate

Abstract

SAPHO syndrome, characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis is rare compared to other spondyloarthropathies. It is also difficult to diagnose, and treatment methods have not yet been fully identified. Approximately 72% of patients are diagnosed with at least one other disease before a final diagnosis of SAPHO syndrome. In addition, SAPHO syndrome is subject to a delayed diagnosis period of 4.5 to 9.1 years. Medications such as non-steroidal anti-inflammatory drugs, disease-modifying anti-rheumatic drugs, and tumor necrosis factor inhibitors are used in treatment of SAPHO syndrome. Bisphosphonate is also used for refractory SAPHO syndrome; however, most reports on this relate to intravenous injection of medication. The authors experienced and subsequently reported on a case involving a patient with SAPHO syndrome accompanied by fracture and infection of the left second finger who was treated with the oral biphosphonate, alendronate.

REFERENCES

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Figure 1.
Pustulosis in different size and shape were seen in her 2nd finger (A) and disappearance of pustule after treatment with 2 months of oral alendronate (B).
jrd-22-313f1.tif
Figure 2.
Bone scan showed increased uptake on the bilateral sternoclavicular joint, manubriosternal junction, both inferior sacroiliac joint, both knees.
jrd-22-313f2.tif
Figure 3.
Magnetic resonance imaging of sagittal (A) and coronal (B) fat suppressed T2 weighted image show bone marrow edema at manubriosternal junction (arrows).
jrd-22-313f3.tif
Table 1.
Summary of reports of patients with SAPHO treated with oral alendronate
Author/year Gender Age (yr) Disease duration (mo) Skin involvement Osteoarticular improvement Time to sustained response (mo)
Fioravanti et al. [9] (2008) F 20 16 Yes Yes 3
Ichikawa et al. [10] (2009) F 52 18 Yes Yes 2
Song et al. [11] (2014) M 42 NA Yes Yes NA
Our study F 54 12 Yes Yes 2

F: female, M: male, NA: not available.

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