Department of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical College, Wielicka 265, 30-663, Krakow, Poland.
Department of Medical Genetics, Institute of Pediatrics, Jagiellonian University Medical College, Krakow, Poland.
Ital J Pediatr. 2018 Aug 15;44(1):92. doi: 10.1186/s13052-018-0526-5.
It has been shown that approximately half of survivors of childhood acute lymphoblastic leukemia (ALL) have symptomatic late effects (LE) that may be severe or life-threatening. The aim of our study was to assess the health status of childhood ALL survivors after over 10 years of follow-up and to assess its relationships with gene polymorphisms, numbers and types of LEs, as well as with intensity of chemotherapy and cranial radiotherapy (CRT).
We conducted a telephone survey in 125 ALL survivors (median time from completion of treatment was 12 years) and compared the results with those obtained in our previous study. Most of the patients were followed-up by local providers.
The prevalence of LEs of approximately 50% was similar in both study groups. More than one LE was found in almost 25% of patients. Endocrine LEs were less frequent than in our previous study (44% vs 22%), probably due to underdiagnosis. The prevalence of hepatitis B/C decreased from 30%/50 to 18% (counted together), and prevalence of neurologic LEs decreased from 18 to 6%. The increase in the rate of second malignancies was not significant (2% vs. 3%). Sixty four percent of patients continued their education at the time of the study. Approximately 51% of ALL survivors who have completed their education by the time of the study had no permanent employment, including 4 mothers of infants and 3 persons qualified for a disability living allowance. These employment problems may have been due to cognitive impairment. The offspring of the ALL survivors included 11 children, all of them healthy. Further analysis showed higher prevalence of hepatitis in patients treated with CRT (p = 0.0001). Genetic studies revealed higher prevalence of hepatitis in patients homozygous for the rs9939609A variant of the FTO gene compared with other patients (p = 0.03). Moreover, wild-type rs1137101 polymorphism (Q223R) of the and leptin receptor gene was more frequent in patients with psychological LEs (p = 0.03).
The prevalence of LEs in ALL survivors is of key importance. The transition of childhood ALL survivors from pediatric to adult care should be urgently improved to maintain continued follow-up provide high-quality care.
Bioethics Committee of the Jagiellonian University approved the study protocol. Registration number: KBET/113/B/2006.
研究表明，约有一半的儿童急性淋巴细胞白血病（ALL）幸存者存在症状性晚期效应（LE），这些效应可能严重或危及生命。我们的研究目的是评估儿童 ALL 幸存者在超过 10 年的随访后的健康状况，并评估其与基因多态性、LE 的数量和类型以及化疗和颅放疗（CRT）强度的关系。
我们对 125 名 ALL 幸存者（完成治疗的中位时间为 12 年）进行了电话调查，并将结果与我们之前的研究进行了比较。大多数患者由当地的医疗服务提供者进行随访。
两组中约 50%的患者存在 LE，这一比例相似。近 25%的患者存在一种以上的 LE。与我们之前的研究相比，内分泌 LE 的发生率较低（44%对 22%），这可能是由于诊断不足所致。乙型肝炎/丙型肝炎的患病率从 30%/50%降至 18%（合并计算），神经 LE 的患病率从 18%降至 6%。第二恶性肿瘤的发生率没有显著增加（2%对 3%）。64%的患者在研究时仍在接受教育。在研究时已完成学业的 ALL 幸存者中，约有 51%没有永久性工作，包括 4 名婴儿的母亲和 3 名有资格领取残疾生活津贴的人。这些就业问题可能是由于认知障碍所致。ALL 幸存者的后代包括 11 名儿童，他们都很健康。进一步的分析显示，接受 CRT 治疗的患者中肝炎的患病率更高（p=0.0001）。遗传研究显示，与其他患者相比，FTO 基因 rs9939609A 变异的纯合子患者中肝炎的患病率更高（p=0.03）。此外，瘦素受体基因的野生型 rs1137101 多态性（Q223R）在存在心理 LE 的患者中更为常见（p=0.03）。
ALL 幸存者 LE 的患病率非常重要。应紧急改善儿童 ALL 幸存者从儿科到成人护理的过渡，以维持持续的随访并提供高质量的护理。
雅盖隆大学生物伦理委员会批准了研究方案。注册号：KBET/113/B/2006。
