肺和胸膜转移的局部型腱鞘巨细胞瘤:病例报告和文献综述。

IF 0.9 Q3 MEDICINE, GENERAL & INTERNAL Journal of Medical Case Reports Pub Date : 2024-09-30 DOI:10.1186/s13256-024-04768-w
Afshan Ali Shaik, Manoj Kumar Panigrahi, Mahismita Patro, Vangapandu Sushmita, Pritinanda Mishra
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引用次数: 0

摘要

背景:腱鞘巨细胞瘤腱鞘巨细胞瘤是一种罕见的关节滑膜、滑囊或腱鞘软组织肿瘤。根据生长方式可分为局部型和弥漫型。局部型腱鞘巨细胞瘤通常是良性的,切除治疗效果良好。弥漫型腱鞘巨细胞瘤与局部型腱鞘巨细胞瘤相反,可破坏骨和软骨,并经常出现局部复发和远处转移。局部型腱鞘巨细胞瘤很少向远处器官转移。在此,我们报告了一例出现肺转移的局部型腱鞘巨细胞瘤,并系统回顾了相关文献:一名 55 岁的亚洲男性因干咳、右侧胸痛和进行性呼吸困难就诊 1 个月。发病前 18 个月,他曾接受过右手食指无痛肿物切除术。肿物在切除后 3 个月内复发,活检结果提示为巨细胞瘤。鉴于怀疑是巨细胞瘤,医生对病灶进行了广泛切除,切除活检结果与腱鞘巨细胞瘤(局部型)的诊断一致。入院时,患者呼吸急促,右侧呼吸音消失。胸片显示右侧胸腔积液,左侧中区有同质不透明。胸部和腹部对比增强计算机断层扫描显示右侧大量胸腔积液和双侧多发分叶状异质性增强胸膜肿块,肿块内部有钙化区。胸腔积液分析显示有渗出物,细胞学检查未发现恶性细胞。肺活检显示破骨细胞样巨细胞和单核纺锤形细胞,有出血和坏死区域,提示腱鞘巨细胞瘤转移。最终诊断为右手食指局部型腱鞘巨细胞瘤,并向肺部和胸膜转移。患者在接受三个周期的地诺单抗注射后因病情进展而去世:结论:肺转移在局部腱鞘巨细胞瘤患者中极为罕见。结论:在局部腱鞘巨细胞瘤患者中,肺转移极为罕见,肺转移患者的生存率通常较低。有必要对局部型腱鞘巨细胞瘤患者进行密切随访,以便及早发现胸肺部并发症。
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Localized type tenosynovial giant cell tumor with metastases to lungs and pleura: a case report and literature review.

Background: 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.

Case presentation: 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.

Conclusion: 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.

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来源期刊
Journal of Medical Case Reports
Journal of Medical Case Reports Medicine-Medicine (all)
CiteScore
1.50
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0.00%
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436
期刊介绍: JMCR is an open access, peer-reviewed online journal that will consider any original case report that expands the field of general medical knowledge. Reports should show one of the following: 1. Unreported or unusual side effects or adverse interactions involving medications 2. Unexpected or unusual presentations of a disease 3. New associations or variations in disease processes 4. Presentations, diagnoses and/or management of new and emerging diseases 5. An unexpected association between diseases or symptoms 6. An unexpected event in the course of observing or treating a patient 7. Findings that shed new light on the possible pathogenesis of a disease or an adverse effect
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