• The Journal of the Korean bone and joint tumor society
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• v.9 no.2
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• pp.206-211
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• 2003

Palmoplantar pustulosis may be associated with peculiar bone and joint conditions. Initially restricted to the anterior chest wall, these conditions were later found to involve peripheral bones and joints. The same aseptic pseudoinfectious bone and joint lesions were also found associated with severe acne. In 1987, Kahn et al suggested this acronym to describe a number of reports of peculiar bone, joint, and skin lesions published over the last 25 years. SAPHO sydrome stands for synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome. We report one case of SAPHO syndrome: A 27-year-old female presented left clavicular and right sacroiliac joint pain with hyperostosis and pustulosis palmaris.

• Journal of Korean Neurosurgical Society
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• v.30 no.11
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• pp.1320-1323
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• 2001

SAPHO syndrome is an acronym for an increasingly recognized syndrome of synovitis, acne, pustulosis, hyperostosis, and osteitis. Most of the previously reported case are from Japan and Europe. The authors report a case of SAPHO syndrome in Korean female who presented with extrasternal neurologic symptoms. A 60-year-old female with thoracic and right chest wall pain presented with the sternocostoclavicular hyperostosis, and recurrent aseptic osteitis. Previously, she had hystrectomy and thyroidectomy due to uterine malignancy 25 years ago. Also, she started to take medications for palm and sole pustulosis 7 days prior to admission. Through evaluation of clinical, radiological, serological studies, studies was done along with bone needle biopsy for the biological reassessment. The hyperostosis was found in the pedicles of thoracic 8, 9, 10 vertebrae and sternoclavicular joint. Radio-isotope bone scan showed an accumulation of tracer in thoracic vertebra. The bone biopsy from these site showed increased osseous turnover, thickening of trabeculae accompanied by mild acculuation of granulation tissue and round cell infiltration, compatible with mild chronic inflammation with marrow fibrosis. The patient showed good response to conservative management. The authors report a case of SAPHO syndrome with thoracic radiculopathy. It is considered that SAPHO syndrome is related to spondyloarthropathy, and appears to have benign disease process with good prognosis.

• The Korean Journal of Nuclear Medicine
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• v.36 no.4
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• pp.255-260
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• 2002

Purpose: SAPHO syndrome is well known to various disease entities including synovitis, acne, pustulosis, hyperostosis and polyarthritis. The purpose of this study is to evaluate sicntigraphic findings and to compare with radiologic findings in SAPHO syndrome. Materials and Methods: Five patients (M:F=5.0, Age $22.8{\pm}4.78$ yrs) with SAPHO syndrome were enrolled in our study. All patients underwent whole-body bone scintigraphy with intravenous administration of 740 MBq of Tc-99m MDP. Among them, two patients were additionally perfomed SPECT of the spine to evaluate the location and extort of spinal lesion. Results: All patients were demonstrated abnormal increased uptakes in sternoclavicular joint (SC), sacroiliac joint (SI), and small joints of both hands. Among them, three patients were bilateral involvement (3/5) and two were unilateral (2/5) in SC. involvement of SI showed bilateral in four patients (4/5) and unilateral in one (1/5). SPECT images demonstrate that the lesion sites of the lumbar spine are more likely facet joints than vertebral bodies or pedicles. Conclusion: As SAPHO syndrome is the disease entity involved polyarticular joints with various dermatologic manifestations, the bone scintigraphy may be a very useful method to evaluate the location and extent of joint involvement, and to avoid inadequate surgical management or ineffective antibiotic treatment.

• Journal of Yeungnam Medical Science
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• v.30 no.1
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• pp.55-57
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• 2013

Synovitis acne pustulosis hyperostosis osteitis (SAPHO) syndrome is a rare disease that involves the skin, bones and joints. It is thought to be caused by infection with low-toxicity bacteria and to be the result of reactive infectious osteitis. However, this hypothesis has not yet been clearly established. New SAPHO syndrome treatment methods are needed because the disease does not respond to treatment in many cases. In this paper, a case is reported of SAPHO syndrome with pain in the acromioclavicular joint and with squamous and pustular macules on the palms and soles. First, the patient was treated with aceclofenac, prednisolon and sulfasalazine for two weeks. However, the symptoms were not relieved, so methotrexate and pamidronate were added to the treatment. Since no improvement was seen after four weeks of treatment, adalimumab was prescribed. The skin lesions were relieved two weeks later, and the bone pain and arthralgia, four weeks later. No recurrence or adverse effects were observed at the 22-week follow-up.

• Investigative Magnetic Resonance Imaging
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• v.18 no.1
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• pp.59-63
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• 2014

A 66-year-old woman was transferred to our hospital due to her right breast cancer. Preoperative breast MRI shows 1.9 cm malignancy on her right breast (cT1N0M0) and incidentally found osteosclerotic change of left coststernoclavicular region. Bone scintigraphy showed hot uptake and the possibility of bone metastasis was not excluded. However, because the bone metastasis is not common in early stage cancer and the costosternoclavicular region is not common site, other possibility should be considered. SAPHO syndrome can be diagnosed even in the absence of dermatosis when there is an axial or appendicular osteitis and hyperostosis, especially in costosternoclavicular region. Though breast imaging specialists are not accustomed to this disease entity, awareness and diagnosis of the SAPHO syndrome can help differentiate bone metastasis.

• Pediatric Infection and Vaccine
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• v.29 no.2
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• pp.96-104
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• 2022

Chronic recurrent multifocal osteomyelitis (CRMO) is an inflammatory bone disorder presenting with sterile osteomyelitis, most often presenting in childhood. Although the etiology is understood incompletely, its association with other auto-inflammatory diseases including inflammatory bowel disease (IBD); psoriasis; Wegener's disease; arthritis; and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome suggests that dysregulated innate immunity may play an important role in the pathogenesis. We report a case of a 13-year-old boy with CRMO associated with Crohn's disease (CD) successfully treated with infliximab after failure of non-steroidal anti-inflammatory drug (NSAID) treatment. He initially was diagnosed with CRMO based on symmetric and aseptic bone lesions with no fever, lack of response to antibiotic treatment, vertebral involvement, and normal blood cell counts. Despite five months of NSAID treatment, his musculoskeletal symptoms were aggravated, and he developed gastrointestinal symptoms. Finally, he was diagnosed with CRMO associated with CD. Due to the severity of symptoms, infliximab was initiated and produced symptom improvement. This case supports infliximab as another choice for treatment of bowel symptoms in addition to the bone and joint symptoms of CRMO when other first-line treatments are ineffective.