Outcome of childhood ALK-positive anaplastic large cell lymphoma relapses: Real-life experience of the French Society of Pediatric Oncology (SFCE) cohort of 75 French children

IF 2.4 3区医学 Q2 HEMATOLOGY

Pediatric Blood & Cancer Pub Date : 2024-10-21 DOI:10.1002/pbc.31397

Victor Pereira, Maël Barthoulot, Nathalie Aladjidi, Audrey Contet, Jean-Hugues Dalle, Marie Émilie Dourthe, Nathalie Garnier, Bénédicte Bruno, Amaury Leruste, Isabelle Pellier, Matthieu Simonin, Catherine Paillard, Arnauld Verschuur, Stéphane Ducassou, Laurence Lamant, Laurence Brugieres, Marie-Cécile Le Deley, Charlotte Rigaud

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Abstract

Objective

To describe treatments and outcomes of French children treated for relapsed/refractory anaplastic lymphoma kinase-positive anaplastic large cell lymphoma (ALK+ ALCL).

Methods

We conducted the analysis of a series of 75 French children treated for a first relapsed/refractory ALK+ ALCL between 1999 and 2017.

Results

The median time to first relapse was 8.1 months from initial diagnosis (2.9 after end of treatment), with 12 relapses during frontline treatment or within 1 month of the end of treatment. Treatment of the first relapse varied according to the period of time and risk factors: 48 received multiagent chemotherapy, including 21 and 19 consolidated with allogeneic stem cell transplantation (SCT) and autologous-SCT, respectively. Twenty-one patients received weekly vinblastine, and six received ALK inhibitors (ALKi). Overall, 64/75 patients reached a second complete remission (CR2). Eight out of 11 patients who did not reach CR2 died and the other three were rescued with ALKi, vinblastine, and nivolumab. With a median follow-up of 8.2 years, 60 patients are alive, 43 in CR2, 15 in CR3, two in CR4; and 15 patients died, six from toxicity and nine from disease progression. The 5-year event-free survival and overall survival after first relapse were 51.7% (95% confidence interval [CI]: 39.6%–62.6%) and 80.7% (95% CI: 69.6%–88.1%), respectively. Time to relapse greater than 12 months from initial diagnosis was proven to be a prognostic factor in relapsed/refractory ALK+ ALCL.

Conclusion

In relapsed ALK+ ALCL, high survival rate can be reached with various therapeutic strategies. The main challenge remains to prevent subsequent relapses, and to lower long-term morbidity.

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儿童ALK阳性无性大细胞淋巴瘤复发的结果：法国儿科肿瘤学会（SFCE）队列中75名法国儿童的真实经历。

目的描述治疗复发/难治性无细胞大细胞淋巴瘤（ALK+ ALCL）的法国儿童的治疗方法和结果：我们对1999年至2017年间接受首次复发/难治性ALK+ ALCL治疗的75名法国儿童进行了系列分析：首次复发的中位时间为初诊后8.1个月（治疗结束后2.9个月），其中12例在一线治疗期间或治疗结束后1个月内复发。首次复发的治疗因时间和风险因素而异：48人接受了多药化疗，其中21人和19人分别接受了异基因干细胞移植（SCT）和自体干细胞移植。21名患者每周接受一次长春新碱治疗，6名患者接受ALK抑制剂（ALKi）治疗。总的来说，64/75名患者达到了第二次完全缓解（CR2）。在未达到CR2的11名患者中，有8名患者死亡，另外3名患者经ALKi、长春新碱和尼伐单抗治疗后获救。中位随访时间为8.2年，60名患者存活，其中43人达到CR2，15人达到CR3，2人达到CR4；15名患者死亡，其中6人死于毒性，9人死于疾病进展。首次复发后的5年无事件生存率和总生存率分别为51.7%（95%置信区间[CI]：39.6%-62.6%）和80.7%（95%置信区间[CI]：69.6%-88.1%）。复发时间超过初始诊断后12个月被证明是复发/难治性ALK+ ALCL的预后因素：结论：对于复发的ALK+ ALCL，各种治疗策略都能达到较高的生存率。结论：对于复发的 ALK+ ALCLL，各种治疗策略都能达到较高的存活率，主要的挑战仍然是如何预防后续复发和降低长期发病率。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Pediatric Blood & Cancer 医学-小儿科

CiteScore

4.90

自引率

9.40%

发文量

546

审稿时长

1.5 months

期刊介绍： Pediatric Blood & Cancer publishes the highest quality manuscripts describing basic and clinical investigations of blood disorders and malignant diseases of childhood including diagnosis, treatment, epidemiology, etiology, biology, and molecular and clinical genetics of these diseases as they affect children, adolescents, and young adults. Pediatric Blood & Cancer will also include studies on such treatment options as hematopoietic stem cell transplantation, immunology, and gene therapy.