Successful treatment of SAPHO syndrome with severe spinal disorder using entercept: a case study

SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, and osteitis) is a rare disease. Presently, there is no treatment guideline for this illness. Several studies suggested entercept, a novel biological agent against tumor necrosis factor-alpha, is effective in treating SAPHO syndrome. We report a case in which the clinical conditions of a middle-aged female patient diagnosed with SAPHO syndrome, with noted spinal disorder, improved significantly after receiving entercept treatment. The patient remained stable after 3-month follow-up.     

Successful treatment of SAPHO syndrome with zoledronic acid

The SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, and osteitis) is a chronic, relapsing, inflammatory condition with skin and osteoarticular manifestations. Its etiology remains unclear, and various treatment regimens with steroids and nonsteroidal antiinflammatory drugs frequently fail to control the disease, while exposing patients to the side effects of these drugs. Because the SAPHO syndrome manifests as a destructive inflammatory bone disease, use of bisphosphonates that possess antiosteoclastic and probably antiinflammatory properties has been suggested to be helpful. To our knowledge, this is the first reported case of successful treatment with zoledronic acid of SAPHO syndrome that was resistant to conventional treatment.     

Successful treatment of SAPHO syndrome with an oral bisphosphonate

The etiology of the synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome remains unclear and treatment with various drugs for SAPHO syndrome often fails. But recently the effectiveness of intravenous bisphosphonates (BPs) for SAPHO syndrome has been reported in many cases and the use of intravenous BPs as first choice drug has been thought to be effective. On the other hand, serious side effects of intravenous BPs were reported in some cases. This is the first reported case of successful treatment of SAPHO syndrome with an oral BPs, the use of which is safer and more practical than intravenous BPs.     

SAPHO syndrome complicated with relapsing polychondritis: A case report

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare chronic inflammatory disease. The main clinical manifestation of SAPHO syndrome is an osteoarthropathy with cutaneous involvement. Relapsing polychondritis (RP) characterized by chronic inflammation and cartilage degeneration is a rare systematic autoimmune disease. Here we report a RP case in a SAPHO syndrome patient, in which auricularitis happened 10 years after the diagnosed as SAPHO syndrome. Tofacitinib treatment can alleviate the symptoms.     

Crohn's disease and the SAPHO syndrome during treatment with infliximab: a case report and review of literature

We report the case of a 40 year-old Turkish woman with severe ileal Crohn's disease associated with palmoplantar pustulosis and aseptic spondylitis, also known as the SAPHO syndrome. However, in the present case, the syndrome developed three months after starting infliximab (Remicade) therapy, which is generally used to treat the SAPHO syndrome. This case-report discusses the unclear relationship between Crohn's disease, the appearance of a SAPHO syndrome and treatment with infliximab.     

Neprhrotic syndrome in a patient with rheumatoid arthritis treated with adalimumab: a case report

The pathogenetic role of TNFalpha in inflammatory diseases has been known for a long time and has modified the therapeutic approach towards this pathology. All over the word, about 400.000 patients have been treated with biological anti-TNF alpha drugs. Particular attention has been taken for the safety of their use. We describe a case of 60 year old man affected by rheumatoid arthritis who has developed nephrotic syndrome with histological pattern typical of systemic lupus erythematosus-like syndrome, without autoantibodies.     

Propionibacterium acnes and SAPHO syndrome: a case report and literature review

OBJECTIVE: To describe the presence of Propionibacterium acnes (P. acnes) in a series of patients with SAPHO syndrome in which a bone biopsy has been carried out and to discuss the results comparing them to the data described in the literature. METHODS: In 6 out of 56 patients with SAPHO syndrome, a bone biopsy from osteitic lesion was carried out. This invasive investigation was performed only in those cases in which it was necessary to clarify the diagnosis. RESULTS: Of the 6 biopsies processed, P. acnes was isolated in only one case. No other infectious agents were identified. CONCLUSION: P. acnes is not often found in bone lesions of SAPHO syndrome. A bone biopsy may represent a procedure useful for corroborating the diagnosis or for excluding other diseases only in specific cases.     

SAPHO综合征一例报道并文献复习

目的 探索ＳＡＰＨＯ综合征的临床特点及诊断方法。方法 报道了一例ＳＡＰＨＯ综合征并对ＳＡＰＨＯ综合征的文献进行复习。结果 本例有手足掌面脓疱疮,双侧锁骨和第一前肋骨炎,骨肥厚。骨病理切片示慢性炎症改变,故确诊为ＳＡＰＨＯ综合征。结论 ＳＡＰＨＯ综合征临床可以见到,应注意和其他疾病鉴别。     

Intracranial manifestations in SAPHO syndrome: the first case report in literature

A 41-year-old woman with SAPHO syndrome presented with numbness of her left arm followed by a generalized seizure. Computed tomography and magnetic resonance imaging of the brain revealed a small ring enhancing lesion in the right parietal lobe with adjacent meningeal thickening and enhancement. Surgical removal and histopathology showed evidence of severe chronic sterile inflammation and no malignant cell. Symptoms recurred and a lesion were again detected on radiological follow-up, but improvement occurred following treatment with antibiotics and biphosphonate, achieving a low inflammatory activity, reduction in CNS lesions and relative clinical well-being. This is a first report in literature about the central nervous system involvement in a patient with SAPHO syndrome.     

Successful treatment of severe nodular scleritis with adalimumab

Ocular features may occur in longstanding rheumatoid arthritis. Among them, scleritis represents the most frequent manifestation of ophthalmologic rheumatoid disease. There are three types of anterior scleritis: diffuse, nodular, and necrotizing with and without inflammation (scleromalacia perforans). Nodular scleritis is the second cause of anterior scleritis representing 20% of all types of scleritis. The conventional treatment of nodular scleritis consists in topical steroids and DMARDS. In severe cases, the therapy with immunosuppressive agents to avoid complications is necessary. We describe a 46-year-old woman presented right nodular scleritis with rheumatoid arthritis. She was treated initially with topical steroids and DMARDs but was unable to received systemic steroids due to left open angle glaucoma in the opposite eye. After an ocular exacerbation, we initiated adalimumab with complete resolution of nodular scleritis. This is the first report of adalimumab in the treatment of nodular scleritis. Adalimumab could be a good alternative in patients with severe nodular scleritis who fail to have conventional treatment with DMARDs and unable to receive systemic steroids.     

血浆置换成功治疗毒蕈中毒性肝病1例

患者男性，47岁，无慢性肝炎病史，无长期大量饮酒史，近期无服用药物史。2005年11月13日与其他4人进食野蘑菇，10h后均出现呕吐、腹泻等胃肠道中毒症状。其中2人次日出现尿黄，并逐渐出现皮肤、巩膜黄染。第4天收治入当地医院治疗，诊断为急性胃肠炎、黄疸性肝炎。肝功能检查：总胆红素（TBil）289μmol／L，直接胆红素（DBil）211．9μmol／L，丙氨酸氨基转移酶（ALT）449U／L，天冬氨酸氨基转移酶（AST）200U／L。电解质和肾功能均正常。经输液治疗，胃肠道症状好转出院。第6日出现头晕、嗜睡，并逐渐出现意识障碍（肝性脑病）。2005年11月20日收治入我科。