血管免疫母细胞t细胞淋巴瘤伴有t(8;14)(q24;q11.2)/TCR::MYC易位,表现为肠道浸润。

IF 3 3区 医学 Q2 HEMATOLOGY Annals of Hematology Pub Date : 2024-12-18 DOI:10.1007/s00277-024-06148-2
Satoshi Ichikawa, Hiroki Kato, Naoya Morota, Hiroaki Abe, Akihisa Kawajiri, Kyoko Inokura, Koichi Onodera, Yasushi Onishi, Noriko Fukuhara, Satoko Sato, Fumiyoshi Fujishima, Ryo Ichinohasama, Hideo Harigae
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引用次数: 0

摘要

虽然MYC癌基因(MYC- r)重排在侵袭性b细胞淋巴瘤中经常观察到,但在t细胞恶性肿瘤中极为罕见。一例64岁男性患者因无症状肺粘膜相关淋巴组织结外边缘区淋巴瘤(MALToma)住院数年,因全身状况不佳及低血压入院。血液检查显示血小板减少和血清乳酸脱氢酶水平升高,而计算机断层扫描显示全身性淋巴结病和脾肿大。腹股沟淋巴结活检诊断为血管免疫母细胞t细胞淋巴瘤(AITL)。入院后不久,患者因AITL多发肠浸润而发生自发性肠穿孔出血。虽然进行了化疗,但患者在入院数周后死亡。鉴定出46,XY,t(8;14)(q24;q11.2)核型,荧光原位杂交分析显示MYC和t细胞受体(TCR) α基因存在分裂信号,由此证实TCR::MYC易位。病理解剖分析显示AITL全身浸润,未见MALToma病变。迄今为止,文献中仅报道了少数成熟t细胞淋巴瘤携带MYC-R的病例。据我们所知,这是首次报道的TCR::MYC重排的AITL病例。这种情况可能与化疗的难治性和侵袭性临床病程有关,包括肠道在内的全身浸润。
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Angioimmunoblastic T-cell lymphoma harboring a t(8;14)(q24;q11.2)/TCR::MYC translocation that presented with intestinal infiltration.

Although rearrangement of the MYC oncogene (MYC-R) is frequently observed in aggressive B-cell lymphomas, it is extremely rare in T-cell malignancies. A 64-year-old man who had been under observation for several years because of asymptomatic pulmonary extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALToma) was admitted to our hospital because of poor general condition and hypotension. Blood tests revealed thrombocytopenia and elevated serum lactate dehydrogenase levels, whereas computed tomography revealed systemic lymphadenopathy and splenomegaly. An inguinal lymph node biopsy precipitated a diagnosis of angioimmunoblastic T-cell lymphoma (AITL). Shortly after admission, the patient experienced spontaneous intestinal perforation and hemorrhage caused by multiple intestinal infiltrations of the AITL. Although chemotherapy was administered, the patient died several weeks after admission. A 46,XY,t(8;14)(q24;q11.2) karyotype was identified, and fluorescence in situ hybridization analyses showed split signals for the MYC and T-cell receptor (TCR) alpha genes, by which a TCR::MYC translocation was confirmed. Pathological autopsy analysis revealed systemic infiltration of the AITL and no MALToma lesions. Only a few cases of mature T-cell lymphoma harboring MYC-R have been reported in the literature thus far. To the best of our knowledge, this is the first reported case of AITL with TCR::MYC rearrangement. This condition could be associated with refractoriness to chemotherapy and aggressive clinical course with systemic infiltration that included the intestine.

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来源期刊
Annals of Hematology
Annals of Hematology 医学-血液学
CiteScore
5.60
自引率
2.90%
发文量
304
审稿时长
2 months
期刊介绍: Annals of Hematology covers the whole spectrum of clinical and experimental hematology, hemostaseology, blood transfusion, and related aspects of medical oncology, including diagnosis and treatment of leukemias, lymphatic neoplasias and solid tumors, and transplantation of hematopoietic stem cells. Coverage includes general aspects of oncology, molecular biology and immunology as pertinent to problems of human blood disease. The journal is associated with the German Society for Hematology and Medical Oncology, and the Austrian Society for Hematology and Oncology.
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