Department of Rheumatology, Faculty of Medicine, Dokuz Eylul University, Balçova/İzmir, Turkey.
Rheumatol Int. 2024 Nov;44(11):2301-2313. doi: 10.1007/s00296-023-05491-3. Epub 2023 Oct 27.
This review provides an overview of SAPHO (Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis), a rare autoinflammatory disease that primarily affects bones, skin, and joints. We conducted a search on Medline/PubMed using keywords such as SAPHO syndrome, chronic recurrent multifocal osteitis/osteomyelitis, and related terms. SAPHO syndrome is rare, with a reported frequency of 1 in 10,000 in the Caucasian population. However, the actual incidence of SAPHO syndrome is unknown, and the incidence of the disease is likely higher. The pathogenesis of SAPHO syndrome remains incompletely understood. Current evidence suggests that SAPHO results from a complex interplay between immune dysregulation, genetic susceptibility, and environmental factors. It's not clear if SAPHO syndrome is an autoimmune disease or an autoinflammatory disease, but current evidence suggests that it's more likely an autoinflammatory disease because of things like neutrophil hyperactivity, fewer natural killer (NK) cells, high levels of interleukin (IL)-1, and a good response to treatments that block IL-1. Osteo-articular (OA) involvement is a key clinical feature of SAPHO. It affects the anterior chest wall, axial skeleton, peripheral joints, mandible, long bones of the extremities, and pelvis. Dermatological involvement is a common target in SAPHO, with lesions observed in 60-90% of cases. Common skin lesions include psoriasis and acne, with hidradenitis suppurativa and neutrophilic dermatoses being less commonly seen. Other clinical findings include constitutional symptoms caused by systemic inflammation, such as fever, weight loss, and fatigue. There is no specific laboratory finding for SAPHO syndrome. However, during active disease, there may be an increase in positive acute phase markers, such as erythrocyte sedimentation rate (ESR), C-reactive protein (CRP), complement levels, mild leukocytosis, and thrombocytosis. Diagnosis is crucial for SAPHO syndrome, which lacks a specific diagnostic finding and is often underrecognized. A comprehensive evaluation of a patient's medical history and physical examination is crucial. Treatment options include non-steroidal anti-inflammatory drugs (NSAIDs), corticosteroids, conventional and synthetic disease-modifying agents (cDMARDs and sDMARDs), biological therapies, bisphosphonates, and antibiotics. Biological treatments have emerged as a viable alternative for SAPHO patients who do not respond to conventional treatments.
这篇综述概述了 SAPHO(滑膜炎、痤疮、脓疱病、骨肥厚和骨炎),一种主要影响骨骼、皮肤和关节的罕见自身炎症性疾病。我们使用 SAPHO 综合征、慢性复发性多灶性骨炎/骨髓炎和相关术语等关键词在 Medline/PubMed 上进行了搜索。SAPHO 综合征较为罕见,白种人群中的报告频率为 1/10000。然而,SAPHO 综合征的实际发病率尚不清楚,而且该疾病的发病率可能更高。SAPHO 综合征的发病机制尚不完全清楚。目前的证据表明,SAPHO 是由免疫失调、遗传易感性和环境因素的复杂相互作用引起的。目前尚不清楚 SAPHO 综合征是自身免疫性疾病还是自身炎症性疾病,但目前的证据表明,由于中性粒细胞过度活跃、自然杀伤 (NK) 细胞较少、白细胞介素 (IL)-1 水平较高以及对阻断 IL-1 的治疗反应良好等原因,它更可能是一种自身炎症性疾病。骨-关节 (OA) 受累是 SAPHO 的一个关键临床特征。它影响前胸壁、轴骨骼、外周关节、下颌骨、四肢长骨和骨盆。皮肤受累是 SAPHO 的常见靶点,60-90%的病例可见皮损。常见的皮肤病变包括银屑病和痤疮,较少见的有化脓性汗腺炎和中性粒细胞皮肤病。其他临床发现包括全身炎症引起的全身症状,如发热、体重减轻和疲劳。SAPHO 综合征没有特定的实验室发现。然而,在疾病活动期,可能会出现阳性急性期标志物的增加,如红细胞沉降率 (ESR)、C 反应蛋白 (CRP)、补体水平、轻度白细胞增多和血小板增多。SAPHO 综合征缺乏特定的诊断发现,且常被漏诊,因此诊断至关重要。对患者的病史和体格检查进行全面评估至关重要。治疗选择包括非甾体抗炎药 (NSAIDs)、皮质类固醇、传统和合成疾病修饰药物 (cDMARDs 和 sDMARDs)、生物疗法、双膦酸盐和抗生素。对于对传统治疗无反应的 SAPHO 患者,生物治疗已成为一种可行的替代方案。