■胸出口综合征(TOS)包括由上胸出口的神经血管结构受压引起的疾病(Roos和Owens,1996年[1];比尔格,2014年;库鲁克,2020[3])。根据压缩结构,它被归类为神经学,动脉和静脉TOS。SAPHO综合征(滑膜炎-痤疮-脓疱病-骨肥大-骨炎综合征)是一种病因不明的罕见慢性炎症性疾病。典型的胸锁关节和锁骨受累,由于该区域的骨肥大引起的并发症,在一些SAPHO综合征病例中,有报道会导致锁骨下静脉血栓形成.2015年至2019年期间,488名患者,患有神经疾病,血管或联合TOS在我们部门提出。根据临床和诊断结果,通过经腋路对175例患者进行了手术治疗,包括完整的第一肋骨和/或颈肋骨切除术,臂丛神经松解术,胸交感神经切除术和血管重建,如果有必要(Curuk,2020)。在此期间,只有1例患者出现SAPHO综合征伴锁骨下静脉和神经血管TOS血栓形成.
方法:我们介绍了一位50岁的女性患者,符合SCARE2020标准(Agha等人。,2020[12])患有极为罕见的神经血管TOS和SAPHO综合征的组合,并因左锁骨肥大而导致左锁骨下静脉血栓形成。
结论:与SAPHO综合征相关的进行性骨改变可导致胸廓出口变窄。目前尚不存在避免锁骨关节和锁骨骨肥厚的药物治疗方法。第一肋骨切除是扩大上胸部区域空间的治疗选择。当然,这是一种罕见的疾病,需要更多的长期随访数据。
UNASSIGNED: Thoracic outlet syndrome (TOS) includes disorders caused by compression of the neurovascular structures in the upper thoracic outlet (Roos and Owens, 1996 [1]; Bürger, 2014; Curuk, 2020 [3]). Depending on the compressed structure, it is categorized into neurological, arterial and venous TOS. SAPHO syndrome (synovitis-acne-pustulosis-hyperostosis-osteitis syndrome) is a rare chronic inflammatory disease of unknown etiology. With its typical involvement of sternoclavicular joint and clavicle, complication due to hyperostosis in this region, leading to thrombosis of the subclavian vein have been reported in some cases of SAPHO syndrome. Between 2015 and 2019 488 patients, suffering from neurological, vascular or combined TOS presented at our department. Depending on clinical and diagnostic results surgical therapy was performed in 175 cases via the transaxillary approach, including complete first rib and/or cervical rib resection, neurolysis of plexus brachialis, thoracic sympathectomy and vascular reconstruction if indicated (Curuk, 2020). During this period, only one single patient presented with SAPHO syndrome with thrombosis of the subclavian vein and neurovascular TOS.
METHODS: We present a 50-year-old female patient, in line with the SCARE 2020 criteria (Agha et al., 2020 [12]) suffering from extremely rare combination of neurovascular TOS and SAPHO syndrome with thrombosis of the left subclavian vein due to hyperostosis of the left clavicle.
CONCLUSIONS: Progressive bone changes associated with SAPHO syndrome can lead to narrowing of the thoracic outlet. Pharmacological therapies to avoid the progression of the hyperostosis of the costoclavicular joint and the clavicle do currently not exist. First rib resection is a therapeutic option to widen the space in the upper thoracic region. Surely, it is a rare condition and more long-term follow-up data are required.