Giant radiation-induced cavernous haemangioma before reduced-intensity bone marrow transplantation for acute lymphoblastic leukaemia.
Giant radiation-induced cavernous haemangioma before reduced-intensity bone marrow transplantation for acute lymphoblastic leukaemia.
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急性淋巴细胞白血病低强度骨髓移植前巨大的放射诱发海绵状血管瘤。
DOI：
10.1038/s41409-018-0272-8
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发表时间：
2019
期刊：
影响因子：
--
通讯作者：
Kawano Y.
中科院分区：
文献类型：
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作者：
Saito A;Nishikawa T;Oyoshi T;Nakagawa S;Kodama Y;Yamada A;Kinoshita M;Okamoto Y;Arita K;Moritake H;Kawano Y.
虽然不常进行，但颅照射用于治疗白血病和淋巴瘤，并防止肿瘤浸润到中枢神经系统（CNS）。放射诱导的海绵状血管瘤（RICH）是颅脑照射的晚期并发症之一，约40%的患者在颅脑照射后10年内累积发展[1-3]。RICH不是辐射诱导的继发性肿瘤，而是由辐射诱导的颅内小血管损伤引起的血管疾病[4]。尽管无症状，但在大多数情况下，RICH在成像检查中偶然发现。无症状患者仅接受观察，即使在诊断后也不接受治疗[2，5，6]。造血干细胞移植（HSCT）后发生RICH的病例很少;然而，没有报告在移植前观察到RICH，也没有任何已知的方法来管理HSCT期间发生的RICH [6]。在这里，我们报告的情况下，一个10岁的男孩与第二次复发的急性淋巴细胞白血病（ALL）与一个巨大的30毫米RICH。在预处理之前，患者出现出血和脑积水，需要枕下开颅术切除血管瘤。在开颅手术后约1个月进行了降低强度的预处理，随后进行了来自无关供体的异基因骨髓移植。患者是一名10岁的男孩，父母健康，没有兄弟姐妹。2岁时，他被诊断为B细胞前体ALL（初始检查结果为白色血细胞计数：37，500/µL;染色体G带：46，XY，t（5; 22）（q35; 11.2），der（9; 15）（q10; q10）; CNS病变：阴性）。患者开始接受多药全身化疗（KYCCSG-ALL 02 [参考文献7]）并获得缓解。在5岁时，他在维持治疗期间发生了孤立的CNS复发;因此，使用ALL-REZ BFM 2002方案重新开始缓解诱导治疗[8]，导致第二次缓解。此后，在6.5岁时，患者接受全脑（18 Gy/10 Fr）和全脊柱（15 Gy/10 Fr）放射治疗。在8岁时，计划的治疗以维持缓解结束。10岁时，常规检查显示白色血细胞计数显著增加至44，600/µL;患者被诊断为第二次复发，仅限于骨髓。染色体G-带显示核型为46，XY，t（5; 9; 22）（q35; q34; q11）。2），荧光原位杂交检测显示bcr-abl融合信号，表明患者患有费城染色体阳性ALL。开始治疗前的头颅磁共振成像（MRI）显示直径为11 mm的大RICH（图1a）。患者开始接受全身化疗（EsPhALL方案[参考文献9]）和辅助伊马替尼治疗。在10.9岁早期强化治疗结束时，次要bcr-abl嵌合mRNA水平低于检测阈值，患者被转诊至我院接受同种异体HSCT。
Although performed less frequently, cranial irradiation is used to treat leukaemia and lymphoma and prevent tumour infiltration to the central nervous system (CNS). One of the late complications of cranial irradiation is radiation-induced cavernous haemangioma (RICH), which develops cumulatively over up to 10 years after cranial irradiation in~ 40% patients [1–3]. Rather than radiation-induced secondary neoplasm, RICH is regarded as a vascular disorder caused by radiation-induced damage to small intracranial vessels [4]. Although asymptomatic, RICH is incidentally detected during imaging tests in most cases. Asymptomatic patients are only observed and not treated even after diagnosis [2, 5, 6]. Few cases of RICH developing after haematopoietic stem cell transplantation (HSCT) have been reported; however, there are no reports in which RICH was observed prior to transplantation nor is there any known method to manage RICH that develops during HSCT [6]. Here, we report the case of a 10-year-old boy with second relapse of acute lymphoblastic leukaemia (ALL) with a giant 30-mm RICH. Prior to conditioning, the patient developed haemorrhage and hydrocephalus that required sub-occipital craniotomy to excise the haemangioma. Reduced-intensity conditioning was performed~ 1 month after craniotomy, followed by allogeneic bone marrow transplantation from an unrelated donor.The patient was a 10-year-old boy with healthy parents and no siblings. At age 2 years, he was diagnosed with B cell precursor ALL (initial exam findings of white blood cell count: 37,500/µL; chromosome G-banding: 46, XY, t (5; 22)(q35; 11.2), der (9; 15)(q10; q10); CNS lesions: negative). He was started on multi-agent systemic chemotherapy (KYCCSG-ALL02 [ref. 7]) and achieved remission. At age 5 year, he experienced an isolated CNS relapse during maintenance therapy; thus, remission-induction therapy was restarted using the ALL-REZ BFM 2002 protocol [8], resulting in a second remission. Thereafter, at age 6.5 years, the patient received radiation therapy to the whole brain (18Gy/10 Fr) and whole spine (15Gy/10 Fr). At age 8 years, the planned treatment was ended with maintained remission. At age 10 years, routine examination revealed a significant increase in white blood cell count at 44,600/µL; the patient was diagnosed with second relapse isolated to the bone marrow. Chromosome G-banding demonstrated a karyotype of 46, XY, t (5; 9; 22)(q35; q34; q11. 2), and fluorescence in situ hybridisation testing revealed a bcr-abl fusion signal, indicating that the patient had Philadelphia chromosome-positive ALL. Cranial magnetic resonance imaging (MRI) prior to starting the treatment revealed a large RICH measuring 11mm in diameter (Fig. 1 a). The patients were started on systemic chemotherapy (EsPhALL regimen [ref. 9]) with adjuvant imatinib. At the end of early intensification therapy at age 10.9 years, the levels of minor bcr-abl chimeric mRNA were below the detection threshold, and the patient was referred to our hospital for allogeneic HSCT.