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皮下脂膜炎样 T 细胞淋巴瘤的遗传特征和临床特征。

Genetic profiles and clinical features in subcutaneous panniculitis-like T-cell lymphomas.

机构信息

School of Medicine and Health Sciences, College of Medicine, University of Tsukuba, Tsukuba, Japan.

Department of Hematology, University of Tsukuba Hospital, Tsukuba, Japan.

出版信息

Cancer Sci. 2024 Nov;115(11):3788-3794. doi: 10.1111/cas.16345. Epub 2024 Sep 17.

DOI:10.1111/cas.16345
PMID:39288772
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11531942/
Abstract

Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is a rare peripheral T-cell lymphoma characterized by cutaneous lesions and immunologic manifestations. The five-year survival rate of SPTCL has been reported to be over 80%, indicating a favorable prognosis. Recent studies have uncovered recurrent germline variants in HAVCR2, encoding an immunomodulator. In this study, we integrated whole-exome sequencing data from 60 samples collected from 36 SPTCL patients, encompassing six patients of our cohort and 30 patients of publicly available data. We identified 138 somatic mutations in skin tumors of 24 patients and HAVCR2 germline mutations in 23 of 29 patients. HAVCR2 p.Tyr82Cys mutations were identified in four of six Japanese patients. During the clinical courses of four patients, cyclophosphamide, hydroxydaunomycin, vincristine, and prednisone were administered to all patients, but it resulted in incomplete responses in all four patients. However, disease conditions of all patients remained stable with additional treatment, including autologous peripheral blood stem cell transplantation. Over a 7.5-year median follow-up, one patient developed autoimmune-related diseases, while one developed other hematological malignancy, resulting in death. To our knowledge, this is the first report of recurrent HAVCR2 germline mutations in Japanese patients, suggesting the necessity for long-term follow-up.

摘要

皮下脂膜炎样 T 细胞淋巴瘤(SPTCL)是一种罕见的外周 T 细胞淋巴瘤,其特征为皮肤损伤和免疫表现。SPTCL 的五年生存率已超过 80%,提示预后良好。最近的研究揭示了编码免疫调节剂的 HAVCR2 中存在反复出现的种系变异。在这项研究中,我们整合了 36 名 SPTCL 患者的 60 个样本的全外显子组测序数据,其中包括我们队列中的 6 名患者和公开数据中的 30 名患者。我们在 24 名患者的皮肤肿瘤中鉴定出 138 个体细胞突变和 29 名患者中的 23 个 HAVCR2 种系突变。在 6 名日本患者中发现了 4 名 HAVCR2 p.Tyr82Cys 突变。在 4 名患者的临床病程中,所有患者均接受了环磷酰胺、羟基柔红霉素、长春新碱和泼尼松治疗,但所有 4 名患者均未完全缓解。然而,所有患者的病情均保持稳定,接受了包括自体外周血干细胞移植在内的额外治疗。在 7.5 年的中位随访中,1 名患者出现了自身免疫相关疾病,1 名患者发生了其他血液系统恶性肿瘤,导致死亡。据我们所知,这是日本患者中首次报道 HAVCR2 种系突变的复发性,提示需要长期随访。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/0ea2e7376094/CAS-115-3788-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/0b40ef5205a8/CAS-115-3788-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/17fba6979ede/CAS-115-3788-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/0ea2e7376094/CAS-115-3788-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/0b40ef5205a8/CAS-115-3788-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/17fba6979ede/CAS-115-3788-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e8f6/11531942/0ea2e7376094/CAS-115-3788-g002.jpg

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本文引用的文献

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Few and far between: clinical management of rare extranodal subtypes of mature T-cell and NK-cell lymphomas.寥寥无几:罕见结外成熟 T 细胞和 NK 细胞淋巴瘤的临床处理方法。
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Frequent germline mutations of in sporadic subcutaneous panniculitis-like T-cell lymphoma.在散发的皮下脂膜炎样 T 细胞淋巴瘤中,经常出现 的种系突变。
Blood Adv. 2019 Feb 26;3(4):588-595. doi: 10.1182/bloodadvances.2018028340.
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Germline HAVCR2 mutations altering TIM-3 characterize subcutaneous panniculitis-like T cell lymphomas with hemophagocytic lymphohistiocytic syndrome.胚系 HAVCR2 突变改变 TIM-3 特征性表现为伴有噬血细胞性淋巴组织细胞增生症的皮下脂膜炎样 T 细胞淋巴瘤。
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