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病例报告:一名儿童霍奇金淋巴瘤长期幸存者发生肺滑膜肉瘤。

Case report: Pulmonary synovial sarcoma in a long-term survivor of childhood Hodgkin lymphoma.

作者信息

Shilo Konstantin, Kneuertz Peter J, Liebner David, Chen Wei

机构信息

Department of Pathology, The Ohio State University Wexner Medical Center, Columbus, OH, United States.

Division of Thoracic Surgery, The Ohio State University Wexner Medical Center, Columbus, OH, United States.

出版信息

Front Oncol. 2023 Feb 24;13:1096160. doi: 10.3389/fonc.2023.1096160. eCollection 2023.

Abstract

Solid organ malignancies have been reported in survivors of Hodgkin lymphoma treated with chemoradiation; however, to the best of our knowledge no cases of pulmonary synovial sarcoma have been documented in the literature in this cohort. We herein provide a detailed description of synovial sarcoma occurring in the lung of a long-term survivor of childhood Hodgkin lymphoma. A 29-year-old female never smoker with past medical history of Hodgkin lymphoma diagnosed at the age of 7 years and treated with chemotherapy and radiation therapy was admitted for management of pneumothorax. Wedge lung resection of an ulcerated subpleural nodule revealed a malignant spindle cell tumor that based on light microscopic and immunohistochemical features was classified as monophasic synovial sarcoma. The diagnosis was further confirmed by identification of SS18 (SYT) rearrangement by fluorescence hybridization and SS18-SSX1 gene fusion by RNA sequencing. The case documents a rare occurrence of synovial sarcoma in a long-term survivor of childhood Hodgkin lymphoma. While comprising a typical genetic profile for synovial sarcoma, the tumor had unusual histological features such as cystic and low-grade morphology. The case suggests that synovial sarcoma falls within an expanding spectrum of secondary malignancies following prior treatment of Hodgkin lymphoma.

摘要

实体器官恶性肿瘤在接受放化疗的霍奇金淋巴瘤幸存者中已有报道;然而,据我们所知,该队列的文献中尚无肺滑膜肉瘤的病例记录。在此,我们详细描述了一名儿童霍奇金淋巴瘤长期幸存者肺部发生的滑膜肉瘤。一名29岁从不吸烟的女性,有7岁时被诊断为霍奇金淋巴瘤并接受化疗和放疗的病史,因气胸入院治疗。对一个溃疡的胸膜下结节进行楔形肺切除,发现一个恶性梭形细胞瘤,根据光镜和免疫组化特征,其被分类为单相滑膜肉瘤。通过荧光杂交鉴定出SS18(SYT)重排以及通过RNA测序鉴定出SS18-SSX1基因融合,进一步证实了诊断。该病例记录了儿童霍奇金淋巴瘤长期幸存者中罕见的滑膜肉瘤发生情况。虽然该肿瘤具有滑膜肉瘤的典型基因特征,但具有不寻常的组织学特征,如囊性和低级别形态。该病例表明,滑膜肉瘤属于霍奇金淋巴瘤先前治疗后继发性恶性肿瘤不断扩大的范围。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a09a/9999020/4805e54761f5/fonc-13-1096160-g001.jpg

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