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一名成人急性淋巴细胞白血病患者在诊断后5年及同种异体移植后4年出现晚期睾丸复发——1例罕见病例

A Late Testicular Relapse in an Adult with Acute Lymphoblastic Leukemia, 5 Years after the Diagnosis and 4 Years after Allotransplant-A Rare Case.

作者信息

Epure Andrei, Pop C Florin, Juravle Ciprian, Grosu Florin, Naicu Carmen

机构信息

Department of Radiology, Sibiu, County Emergency Hospital, 540245 Sibiu, Romania.

Department of Surgery, Jules Bordet Institute, Université Libre de Bruxelles, 90 Rue Meylemeersch, 1070 Brussels, Belgium.

出版信息

Diagnostics (Basel). 2024 Apr 23;14(9):875. doi: 10.3390/diagnostics14090875.

DOI:10.3390/diagnostics14090875
PMID:38732290
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11083869/
Abstract

Acute lymphoblastic leukemia (ALL) is a malignant disorder of lymphoid progenitor cells that affects both pediatric and adult populations. Although isolated testicular or any other organ recurrence can occur in the pediatric population, it is rare in adults. We present images for an atypical case of the late testicular recurrence of acute lymphoblastic leukemia in a 56-year-old man previously diagnosed with ALL pro-T who was in remission following polychemotherapy (GMALL 2013 protocol) and the allotransplantation of peripheral blood stem cells from a related donor. Five years later (2022), the unilateral testicular relapse of ALL was suspected by imaging and diagnosed by immunophenotyping from sperm fluid infiltrated with atypical cells with an immunophenotype concordant with that of the underlying disease (ALL T). Bone marrow aspiration and biopsy showed no evidence of systemic leukemia relapse. Testicular ablation or chemotherapy and irradiation were considered. Given the strictly testicular relapse, orchiectomy would have been useful, but given the abdominal adenopathy, a chemotherapy course with HyperCVAD Block A was first required. Testicular relapse can occur at any age, and the recognition of this is important as it may be the first manifestation of systemic relapse.

摘要

急性淋巴细胞白血病(ALL)是一种影响儿童和成人的淋巴祖细胞恶性疾病。虽然在儿童群体中可发生孤立性睾丸或其他任何器官复发,但在成人中罕见。我们展示了一名56岁男性急性淋巴细胞白血病晚期睾丸复发的非典型病例的影像资料,该男性此前被诊断为ALL pro-T,在接受多药化疗(GMALL 2013方案)及来自相关供体的外周血干细胞同种异体移植后处于缓解期。五年后(2022年),通过影像学怀疑ALL单侧睾丸复发,并通过对浸润有非典型细胞的精液进行免疫表型分析确诊,其免疫表型与基础疾病(ALL T)一致。骨髓穿刺和活检未显示系统性白血病复发的证据。考虑进行睾丸切除或化疗及放疗。鉴于严格的睾丸复发情况,睾丸切除术可能会有用,但鉴于腹部淋巴结肿大,首先需要进行一个疗程的HyperCVAD A方案化疗。睾丸复发可发生于任何年龄,认识到这一点很重要,因为它可能是系统性复发的首发表现。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/7edde9a42425/diagnostics-14-00875-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/c0dd4f78d783/diagnostics-14-00875-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/03fac575197c/diagnostics-14-00875-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/6f2f5128819c/diagnostics-14-00875-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/7edde9a42425/diagnostics-14-00875-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/c0dd4f78d783/diagnostics-14-00875-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/03fac575197c/diagnostics-14-00875-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/6f2f5128819c/diagnostics-14-00875-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a8/11083869/7edde9a42425/diagnostics-14-00875-g004.jpg

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本文引用的文献

1
Is testicular irradiation necessary for patients with acute lymphoblastic leukemia and testicular relapse?急性淋巴细胞白血病合并睾丸复发的患者是否需要进行睾丸照射?
Pediatr Blood Cancer. 2018 May;65(5):e26977. doi: 10.1002/pbc.26977. Epub 2018 Jan 19.
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Mutational profiling of acute lymphoblastic leukemia with testicular relapse.伴有睾丸复发的急性淋巴细胞白血病的突变谱分析。
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Continued complete remission without systemic therapy for isolated testicular relapse after bone marrow transplantation in a boy with acute lymphoblastic leukemia.
一名急性淋巴细胞白血病男孩在骨髓移植后出现孤立性睾丸复发,未进行全身治疗但持续完全缓解。
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Isolated testicular relapse after bone marrow transplant with total body irradiation and testicular boost in acute lymphoblastic leukemia.急性淋巴细胞白血病患者在接受全身照射和睾丸局部照射的骨髓移植后出现孤立性睾丸复发。
Bone Marrow Transplant. 1998 Aug;22(4):397-9. doi: 10.1038/sj.bmt.1701340.
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Testicular relapse in adult acute lymphocytic leukemia: a case report and literature review.成人急性淋巴细胞白血病睾丸复发:一例报告及文献复习
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Treatment of occult or late overt testicular relapse in children with acute lymphoblastic leukemia: a Pediatric Oncology Group study.儿童急性淋巴细胞白血病隐匿性或晚期显性睾丸复发的治疗:一项儿科肿瘤学组研究
J Clin Oncol. 1992 Apr;10(4):624-30. doi: 10.1200/JCO.1992.10.4.624.