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一例罕见的肩部弥漫性色素沉着绒毛结节性滑膜炎:关节镜下滑膜切除术及辅助放疗的多学科治疗方法

An unusual case of diffuse pigmented villonodular synovitis of the shoulder: A multidisciplinary approach with arthroscopic synovectomy and adjuvant radiotherapy.

作者信息

Serra Tânia Quinás, Morais João, Gonçalves Zico, Agostinho Francisco, Melo Gilberto, Henriques Mónica

机构信息

Department of Radioncology, IPO CFG EPE, Coimbra, Portugal.

Department of Orthopaedics and Traumatology, Centro Hospitalar Tondela Viseu, Viseu, Portugual.

出版信息

Eur J Rheumatol. 2017 Jun;4(2):142-144. doi: 10.5152/eurjrheum.2016.15084. Epub 2017 Feb 24.

Abstract

Pigmented villonodular synovitis (PVNS) is a rare proliferative disorder of the synovial membrane. This condition is usually monoarticular, can be locally destructive, and involves muscles, tendons, bursae, bones, and skin. The most commonly affected joints are the knee and hip, followed by the ankle and shoulder. Patients often present with pain, swelling, and joint effusion; however, the duration of symptoms varies. Total synovectomy is the preferred treatment for PVNS. Subtotal synovectomy is a factor of recurrence, and in diffuse PVNS, total excision is very difficult to achieve. Radiotherapy may have an adjunctive role, particularly in incomplete resection or as a treatment of salvation in recurrent cases. This treatment modality has low toxicity levels and enables satisfactory joint function. This is a case report of a rare case of diffuse PVNS of the shoulder that was treated with partial arthroscopic synovectomy and adjuvant radiotherapy. A 74-year-old male patient presented with gradual onset pain, hemarthrosis, and functional impairment of the right shoulder without previous trauma history. Magnetic resonance imaging of the shoulder demonstrated a diffuse synovial thickening that was compatible with PVNS and rotator cuff destruction. The lesion was partially excised by arthroscopy. The patient underwent adjuvant radiation therapy with a total dose of 40 Gy/20 fractions/4 weeks. At the final follow-up, i.e., 1 month after treatment, the patient had increased shoulder mobility and no pain, with a mild change in cutaneous pigmentation. Radiation therapy is safe and effective in treating and preventing recurrence of diffuse PVNS, particularly after incomplete synovectomy.

摘要

色素沉着绒毛结节性滑膜炎(PVNS)是一种罕见的滑膜增殖性疾病。这种病症通常为单关节性,具有局部破坏性,可累及肌肉、肌腱、滑囊、骨骼和皮肤。最常受累的关节是膝关节和髋关节,其次是踝关节和肩关节。患者常表现为疼痛、肿胀和关节积液;然而,症状持续时间各不相同。全滑膜切除术是PVNS的首选治疗方法。次全滑膜切除术是复发的一个因素,而在弥漫性PVNS中,完全切除很难实现。放射治疗可能具有辅助作用,特别是在不完全切除的情况下或作为复发病例的挽救性治疗。这种治疗方式毒性水平低,能使关节功能令人满意。本文报告了一例罕见的肩部弥漫性PVNS病例,采用关节镜下部分滑膜切除术和辅助放疗进行治疗。一名74岁男性患者,无既往创伤史,逐渐出现右肩部疼痛、关节积血和功能障碍。肩部磁共振成像显示弥漫性滑膜增厚,符合PVNS及肩袖损伤。通过关节镜对病变进行了部分切除。患者接受了辅助放疗,总剂量为40Gy/20次/4周。在最后一次随访时,即治疗后1个月,患者肩部活动度增加且无疼痛,皮肤色素沉着有轻微变化。放射治疗在治疗和预防弥漫性PVNS复发方面是安全有效的,特别是在滑膜切除不完全之后。

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