J Rheum Dis.  2015 Oct;22(5):313-316. 10.4078/jrd.2015.22.5.313.

A Case Report of SAPHO Syndrome Treated with Oral Alendronate

Affiliations
  • 1Division of Rheumatology, Department of Internal Medicine, Inha University Hospital, Inha University School of Medicine, Incheon, Korea. rhksr@inha.ac.kr
  • 2Division of Infectious Diseases, Department of Internal Medicine, Inha University Hospital, Inha University School of Medicine, Incheon, Korea.
  • 3Department of Radiology, Inha University Hospital, Inha University School of Medicine, Incheon, Korea.

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.

Keyword

Acquired hyperostosis syndrome; Therapeutics; Alendronate

MeSH Terms

Acne Vulgaris
Acquired Hyperostosis Syndrome*
Alendronate*
Antirheumatic Agents
Delayed Diagnosis
Diagnosis
Fingers
Humans
Hyperostosis
Injections, Intravenous
Osteitis
Spondylarthropathies
Synovitis
Tumor Necrosis Factor-alpha
Alendronate
Antirheumatic Agents
Tumor Necrosis Factor-alpha

Figure

  • 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).

  • Figure 2. Bone scan showed increased uptake on the bilateral sternoclavicular joint, manubriosternal junction, both inferior sacroiliac joint, both knees.

  • Figure 3. Magnetic resonance imaging of sagittal (A) and coronal (B) fat suppressed T2 weighted image show bone marrow edema at manubriosternal junction (arrows).


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