Ohida H, Curuk C, Prescher H, Stegemann E, Bürger Th
Agaplesion Diakonie Kliniken Kassel gGmbH, Department of Vascular and Endovascular Surgery, Herkulesstraße 34, 34119 Kassel, Germany.
Agaplesion Diakonie Kliniken Kassel gGmbH, Department of Vascular and Endovascular Surgery, Herkulesstraße 34, 34119 Kassel, Germany.
Int J Surg Case Rep. 2021 Mar;80:105710. doi: 10.1016/j.ijscr.2021.105710. Epub 2021 Feb 25.
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.
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.
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.
胸廓出口综合征（TOS）包括由胸廓上口神经血管结构受压引起的疾病（鲁斯和欧文斯，1996 [1]；比尔格，2014；库鲁克，2020 [3]）。根据受压结构的不同，可分为神经型、动脉型和静脉型TOS。滑膜炎-痤疮-脓疱病-骨肥厚-骨炎综合征（SAPHO综合征）是一种病因不明的罕见慢性炎症性疾病。由于其典型地累及胸锁关节和锁骨，在一些SAPHO综合征病例中，曾报道过该区域骨肥厚导致的并发症，进而引起锁骨下静脉血栓形成。2015年至2019年间，488例患有神经型、血管型或混合型TOS的患者到我们科室就诊。根据临床和诊断结果，175例患者通过经腋窝入路进行了手术治疗，包括完整切除第一肋和/或颈肋、臂丛神经松解、胸交感神经切除术以及必要时的血管重建（库鲁克，2020）。在此期间，仅有1例患者同时患有伴有锁骨下静脉血栓形成的SAPHO综合征和神经血管型TOS。
我们报告一名50岁女性患者，符合2020年SCARE标准（阿加等人，2020 [12]），患有极为罕见的神经血管型TOS与SAPHO综合征合并症，因左锁骨骨肥厚导致左锁骨下静脉血栓形成。
与SAPHO综合征相关的进行性骨质改变可导致胸廓出口狭窄。目前尚无避免肋锁关节和锁骨骨肥厚进展的药物治疗方法。切除第一肋是扩大胸廓上部空间的一种治疗选择。当然，这是一种罕见病症，需要更多长期随访数据。
