原发性纵隔精原细胞瘤合并平滑肌肉瘤1例。

Yali Qi, Chi Dong, Wenjun Cui, Lijun Da, Enxi Li, Yating Liu, Feixue Song
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引用次数: 1

摘要

生殖细胞肿瘤是生殖腺最常见的恶性肿瘤,有时在生殖腺以外的部位发现,称为生殖腺外生殖细胞肿瘤(EGCTs)。原发性纵隔生殖细胞肿瘤(pmgct)是一种罕见的前纵隔肿瘤,包括精原细胞瘤和非精原细胞瘤,或以混合形式出现。摘要原发性纵隔精原细胞瘤合并肉瘤是一种极为罕见的临床病理实体。先前的研究表明,原发性纯纵隔精原细胞瘤通常对放化疗敏感,也可能对姑息性切除敏感。由精原细胞瘤和肉瘤组成的混合性生殖细胞瘤的治疗方案尚不清楚。目前文献中仅报道1例原发性纵隔精原细胞瘤合并横纹肌肉瘤,患者受益于化疗作为新辅助治疗。然而,原发纵隔精原细胞瘤合并平滑肌肉瘤的病例尚未见文献记载。在此,我们报告一例18岁的患者,其表现为呼吸困难、直立呼吸和胸痛,胸部CECT扫描显示前纵隔有一大块肿块,部分切除后证实为精原细胞瘤合并平滑肌肉瘤。我们将探讨该病的治疗策略和潜在的分子机制。最后,我们的研究表明,患者受益于化疗单独治疗,或术后联合靶向治疗。同时,BRAF p.G466V、TP53突变、MTOR p.T1977I和FLCN外显子2-5缺失可能是本病潜在的分子机制和致癌驱动因素。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Primary mediastinal seminoma with leiomyosarcoma: a rare case report.

Germ cell tumor is the most common malignant tumor of the gonads, sometimes they are found in locations other than the gonads, called Extra-gonadal Germ cell tumours (EGCTs). Primary mediastinal germ cell tumors (PMGCTs) are a kind of rare neoplasm in the anterior mediastinum, including seminoma and non-seminomatous, or appear as a mixture. Primary mediastinal seminoma mixed with sarcoma is an extremely rare clinicopathologic entity. Previous studies have revealed that primary pure mediastinal seminomas are commonly sensitive to chemoradiotherapy and possibly to palliative excision. The treatment options for mixed germ cell tumor composed of seminoma and sarcoma remain unknown. Only one case of primary mediastinal seminoma with rhabdosarcoma has been reported in the literature up to date and the patient benefited from chemotherapy as the neoadjuvant therapy. However, cases of primary mediastinal seminoma with leiomyosarcoma have not been documented. Herein, we report a case of an 18-year-old patient, who presented with dyspnea, orthopnea, and chest pain, the CECT scan of the chest showed a large mass in the anterior mediastinum, which turned out to be seminoma mixed with leiomyosarcoma after partial excision. We investigate the treatment strategy and potential molecular mechanism of this disease. Finally, our study demonstrated that the patient benefited from the treatment of chemotherapy alone, or combined with target therapy after the operation. Meanwhile, the BRAF p.G466V, TP53 mutations, MTOR p.T1977I and exons 2-5 deletion of FLCN may be potential molecular mechanisms and oncogenic drivers of this disease.

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