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畸胎瘤转变为血管肉瘤:纵隔内的一种蜕变

Teratoma to Angiosarcoma: A Metamorphosis in the Mediastinum.

作者信息

Acharya Sudeep, Pokima Ngowari, Yetiskul Ekrem, Achkar Michel, Grabie Yisroel Y, Khanijo Sameer, Villa Sanchez Manuel, Chalhoub Michel

机构信息

Pulmonary and Critical Care Medicine, Staten Island University Hospital, Staten Island, USA.

Internal Medicine, Staten Island University Hospital, Staten Isand, USA.

出版信息

Cureus. 2024 Jun 17;16(6):e62555. doi: 10.7759/cureus.62555. eCollection 2024 Jun.

DOI:10.7759/cureus.62555
PMID:39022476
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11254467/
Abstract

We describe a rare and remarkable transformation of an immature mediastinal teratoma into high-grade angiosarcoma in a 21-year-old male. Mediastinal teratomas, particularly immature ones, are exceedingly rare, representing a small fraction of germ cell tumors (GCTs). Our case describes the clinical journey of the patient, who initially presented with acute chest pain and was subsequently diagnosed with an immature teratoma following imaging studies and elevated tumor markers. Despite an initial positive response to cisplatin-based chemotherapy, surveillance imaging revealed liver masses, which a biopsy confirmed as angiosarcoma. This transformation underscores the aggressive nature of immature teratomas and the propensity for sarcomatous differentiation, particularly in the mediastinum. The case contributes valuable insight into the management and surveillance of mediastinal non-seminoma germ cell tumors (MNGCT), a subset of GCTs with limited literature. We believe this case is the first in the literature to describe a transformation from an immature teratoma in the mediastinum to a high-grade angiosarcoma.

摘要

我们描述了一名21岁男性中罕见且显著的未成熟纵隔畸胎瘤转变为高级别血管肉瘤的病例。纵隔畸胎瘤,尤其是未成熟型,极为罕见,仅占生殖细胞肿瘤(GCTs)的一小部分。我们的病例描述了该患者的临床病程,患者最初表现为急性胸痛,随后经影像学检查及肿瘤标志物升高被诊断为未成熟畸胎瘤。尽管最初对基于顺铂的化疗有阳性反应,但监测影像学显示肝脏有肿块,活检证实为血管肉瘤。这种转变突显了未成熟畸胎瘤的侵袭性本质以及肉瘤样分化的倾向,尤其是在纵隔部位。该病例为纵隔非精原细胞生殖细胞肿瘤(MNGCT)的管理和监测提供了有价值的见解,MNGCT是GCTs的一个子集,相关文献有限。我们认为该病例是文献中首例描述纵隔未成熟畸胎瘤转变为高级别血管肉瘤的病例。

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1
Teratoma to Angiosarcoma: A Metamorphosis in the Mediastinum.畸胎瘤转变为血管肉瘤:纵隔内的一种蜕变
Cureus. 2024 Jun 17;16(6):e62555. doi: 10.7759/cureus.62555. eCollection 2024 Jun.
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Benign clinical behavior of immature mediastinal teratoma in infancy and childhood: report of two cases and review of the literature.婴幼儿及儿童期未成熟纵隔畸胎瘤的良性临床行为:两例报告并文献复习
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本文引用的文献

1
A case of mediastinal teratoma with malignant transformation into angiosarcoma and relapse with multiple bone metastases that was cured by a multidisciplinary treatment.一例纵隔畸胎瘤恶变为血管肉瘤并伴有多发骨转移复发,经多学科治疗后治愈。
IJU Case Rep. 2022 Nov 11;6(2):93-96. doi: 10.1002/iju5.12551. eCollection 2023 Mar.
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Primary Mediastinal Germ Cell Tumors: A Thorough Literature Review.原发性纵隔生殖细胞肿瘤:全面的文献综述
Biomedicines. 2023 Feb 8;11(2):487. doi: 10.3390/biomedicines11020487.
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Immature ovarian teratoma in childhood: Case report of successful management of a monster mass in a preschool girl.
儿童期未成熟卵巢畸胎瘤:一名学龄前女童巨大肿物成功治疗的病例报告
Ann Med Surg (Lond). 2022 Jul 9;80:104147. doi: 10.1016/j.amsu.2022.104147. eCollection 2022 Aug.
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How to classify, diagnose, treat and follow-up extragonadal germ cell tumors? A systematic review of available evidence.如何对生殖细胞肿瘤进行分类、诊断、治疗和随访?现有证据的系统评价。
World J Urol. 2022 Dec;40(12):2863-2878. doi: 10.1007/s00345-022-04009-z. Epub 2022 May 12.
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The differences between gonadal and extra-gonadal malignant teratomas in both genders and the effects of chemotherapy.两性性腺和性腺外恶性畸胎瘤的差异,以及化疗的影响。
BMC Cancer. 2019 Apr 30;19(1):408. doi: 10.1186/s12885-019-5598-0.
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Pure Immature Teratoma of the Ovary in Adults: Thirty-Year Experience of a Single Tertiary Care Center.成人卵巢纯未成熟畸胎瘤:一家三级医疗中心的30年经验
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Extragonadal teratomas of the adult abdomen and pelvis: a pictorial review.成人腹部和盆腔的性腺外畸胎瘤:影像学综述
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Salvage therapy with high-dose chemotherapy and peripheral blood stem cell transplant in patients with primary mediastinal nonseminomatous germ cell tumors.采用大剂量化疗和外周血干细胞移植治疗原发性纵隔非精原细胞瘤生殖细胞肿瘤患者。
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Teratoma with malignant transformation: a case report with pathological, cytogenetic, and immunohistochemistry analysis.伴有恶性转化的畸胎瘤:一例病例报告及病理、细胞遗传学和免疫组织化学分析
Sarcoma. 2011;2011:450743. doi: 10.1155/2011/450743. Epub 2011 Jun 9.