Department of Pulmonary Medicine and Critical Care, All India Institute of Medical Sciences, Bhubaneswar, 751019, India.
Department of Pathology and Laboratory Medicine, All India Institute of Medical Sciences, Bhubaneswar, India.
J Med Case Rep. 2024 Sep 30;18(1):452. doi: 10.1186/s13256-024-04768-w.
Tenosynovial giant cell tumor is a rare soft tissue tumor of the synovium of joint, bursae, or tendon sheath. It is divided into localized or diffuse types on the basis of the growth pattern. Localized tenosynovial giant cell tumors are usually benign and treated successfully by excision. Diffuse tenosynovial giant cell tumors, in contrast to localized type, can destroy bone and cartilage and are associated with frequent local recurrences and distant metastasis. Localized type tenosynovial giant cell tumors rarely metastasize to distant organs. Here, we report a case of localized tenosynovial giant cell tumor presenting with lung metastases and systematically review literature.
A 55-year-old Asian male presented with a dry cough, right-sided chest pain and progressive dyspnea for 1 month. At 18 months before this presentation, he had undergone excision of a painless swelling on his right index finger. The swelling recurred within 3 months of excision, and a biopsy was then suggestive of a giant cell tumor. Given the suspicion of a giant cell tumor, a wide excision of the lesion was performed and the excisional biopsy was consistent with a diagnosis of tenosynovial giant cell tumor, localized type. At admission to our hospital, the patient had tachypnoea and absent breath sounds on the right side. A chest radiograph showed a right-sided pleural effusion with a homogenous opacity in the left mid-zone. A contrast-enhanced computed tomography of the chest and abdomen showed right massive pleural effusion and bilateral multiple lobulated heterogeneously enhancing pleural-based masses with areas of internal calcification. Pleural fluid analysis revealed an exudate with no malignant cells on cytology. A lung biopsy showed osteoclast-like giant cells and mononuclear spindle cells with areas of hemorrhage and necrosis, suggesting tenosynovial giant cell tumor metastasis. A final diagnosis of localized type tenosynovial giant cell tumor of the right index finger with metastases to the lungs and pleura was made. The patient passed away after receiving three cycles of denosumab injection owing to progressive disease.
Lung metastasis is extremely rare in patients with localized tenosynovial giant cell tumor. The survival is usually poor in patients with lung metastasis. A close follow-up of patients with localized type tenosynovial giant cell tumor is necessary for early detection of pleuropulmonary complications.
腱鞘巨细胞瘤是一种罕见的滑膜组织肿瘤,发生于关节、滑囊或腱鞘。根据生长模式,它分为局限性或弥漫性两种类型。局限性腱鞘巨细胞瘤通常为良性,通过切除可成功治疗。相比之下,弥漫性腱鞘巨细胞瘤可破坏骨骼和软骨,且常局部复发和远处转移。局限性腱鞘巨细胞瘤很少转移至远处器官。在此,我们报告一例局限性腱鞘巨细胞瘤伴肺转移病例,并系统复习文献。
一名 55 岁亚裔男性,因干咳、右侧胸痛和进行性呼吸困难 1 个月就诊。在此次就诊前 18 个月,他曾因右手食指无痛性肿胀接受切除手术。切除后 3 个月内肿胀复发,活检提示为巨细胞瘤。鉴于巨细胞瘤的怀疑,对病变进行了广泛切除,切除活检结果与腱鞘巨细胞瘤、局限性一致。入院时,患者呼吸急促,右侧呼吸音消失。胸部 X 线片显示右侧胸腔积液,左中肺区均匀密度增高。胸部和腹部增强 CT 显示右侧大量胸腔积液和双侧多发性分叶状不均匀强化胸腔基底部肿块,伴内部钙化。胸腔积液分析显示细胞学无恶性细胞的渗出液。肺活检显示破骨样巨细胞和单核梭形细胞,伴出血和坏死区,提示腱鞘巨细胞瘤转移。最终诊断为右手食指局限性腱鞘巨细胞瘤伴肺和胸膜转移。由于疾病进展,患者在接受 3 周期地舒单抗注射后去世。
局限性腱鞘巨细胞瘤患者发生肺转移极为罕见。发生肺转移的患者预后通常较差。对局限性腱鞘巨细胞瘤患者进行密切随访对于早期发现胸膜肺并发症非常必要。