Symptomatic osteonecrosis in French survivors of childhood and adolescent leukemia: a clinical and MRI study of LEA cohort.

IF 1.2 4区 医学 Q4 HEMATOLOGY
Alice Huault, Gérard Michel, Valérie Charon, Kamal Chouklati, Carine Domenech, Pascal Chastagner, Jean-Hugues Dalle, Catherine Paillard, Stéphane Ducassou, Marilyne Poirée, Geneviève Plat, Marie-Dominique Tabone, Justyna Kanold, André Baruchel, Claire Berger, Isabelle Pellier, Dominique Plantaz, Alexandre Theron, Alaa Mustafa, Pascal Auquier, Virginie Gandemer
{"title":"Symptomatic osteonecrosis in French survivors of childhood and adolescent leukemia: a clinical and MRI study of LEA cohort.","authors":"Alice Huault,&nbsp;Gérard Michel,&nbsp;Valérie Charon,&nbsp;Kamal Chouklati,&nbsp;Carine Domenech,&nbsp;Pascal Chastagner,&nbsp;Jean-Hugues Dalle,&nbsp;Catherine Paillard,&nbsp;Stéphane Ducassou,&nbsp;Marilyne Poirée,&nbsp;Geneviève Plat,&nbsp;Marie-Dominique Tabone,&nbsp;Justyna Kanold,&nbsp;André Baruchel,&nbsp;Claire Berger,&nbsp;Isabelle Pellier,&nbsp;Dominique Plantaz,&nbsp;Alexandre Theron,&nbsp;Alaa Mustafa,&nbsp;Pascal Auquier,&nbsp;Virginie Gandemer","doi":"10.1080/08880018.2023.2168810","DOIUrl":null,"url":null,"abstract":"<p><p>Osteonecrosis (ON) is a known complication of acute leukemia (AL) management, affecting 1%-10% of young patients and resulting in long-term morbidity. Widespread access to MRI over the past decade has allowed earlier detection and more accurate assessment. This study investigated clinical and MRI features of the 129 (2.5%) patients with symptomatic ON retrospectively recruited from the French LEA (Leucémies de l'Enfant et de l'Adolescent, or <i>child and adolescent leukemias</i>) cohort (<i>n</i> = 4,973). We analyzed data concerning ON risk factors, multifocal involvement, severe lesions detected by MRI, and patient quality of life (QoL). ON patients tended to be >10 years old at the time of AL diagnosis (odds ratio [OR]: 22.46; <i>p</i> < 10<sup>-6</sup>), female (OR: 1.8; <i>p</i> = 0.002), or treated for relapse (OR: 1.81; <i>p</i> = 0.041). They more frequently suffered from other sequelae (<i>p</i> < 10<sup>-6</sup>). Most necroses involved weight-bearing joints, and they were multifocal in 69% of cases. Double-blinded review of MRIs for 39 patients identified severe lesions in 14, usually in the hips. QoL of adolescents and adults was poor and permanently impacted after onset of ON. In conclusion, age >10 at time of AL diagnosis, female sex, and relapse occurrence were risk factors for multifocal ON; MRI revealed severe ON in a third of the patients considered; and ON was associated with persistently poor QoL affecting multiple domains. Future studies should include prospective data addressing ON management and seek to identify genetic markers for targeted screening enabling early ON detection and treatment.</p>","PeriodicalId":19746,"journal":{"name":"Pediatric Hematology and Oncology","volume":null,"pages":null},"PeriodicalIF":1.2000,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Hematology and Oncology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1080/08880018.2023.2168810","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"HEMATOLOGY","Score":null,"Total":0}
引用次数: 0

Abstract

Osteonecrosis (ON) is a known complication of acute leukemia (AL) management, affecting 1%-10% of young patients and resulting in long-term morbidity. Widespread access to MRI over the past decade has allowed earlier detection and more accurate assessment. This study investigated clinical and MRI features of the 129 (2.5%) patients with symptomatic ON retrospectively recruited from the French LEA (Leucémies de l'Enfant et de l'Adolescent, or child and adolescent leukemias) cohort (n = 4,973). We analyzed data concerning ON risk factors, multifocal involvement, severe lesions detected by MRI, and patient quality of life (QoL). ON patients tended to be >10 years old at the time of AL diagnosis (odds ratio [OR]: 22.46; p < 10-6), female (OR: 1.8; p = 0.002), or treated for relapse (OR: 1.81; p = 0.041). They more frequently suffered from other sequelae (p < 10-6). Most necroses involved weight-bearing joints, and they were multifocal in 69% of cases. Double-blinded review of MRIs for 39 patients identified severe lesions in 14, usually in the hips. QoL of adolescents and adults was poor and permanently impacted after onset of ON. In conclusion, age >10 at time of AL diagnosis, female sex, and relapse occurrence were risk factors for multifocal ON; MRI revealed severe ON in a third of the patients considered; and ON was associated with persistently poor QoL affecting multiple domains. Future studies should include prospective data addressing ON management and seek to identify genetic markers for targeted screening enabling early ON detection and treatment.

法国儿童和青少年白血病幸存者的症状性骨坏死:LEA队列的临床和MRI研究。
骨坏死(ON)是急性白血病(AL)治疗的一种已知并发症,影响1%-10%的年轻患者,并导致长期发病率。在过去的十年中,MRI的广泛使用使得早期发现和更准确的评估成为可能。本研究回顾性调查了129例(2.5%)有症状的ON患者的临床和MRI特征,这些患者来自法国LEA (leucmies de l’enfant et de l’adolescent,或儿童和青少年白血病)队列(n = 4,973)。我们分析了有关ON危险因素、多灶受累、MRI检测到的严重病变和患者生活质量(QoL)的数据。ON患者在AL诊断时倾向于>10岁(优势比[OR]: 22.46;p -6),女性(OR: 1.8;p = 0.002),或治疗复发(or: 1.81;p = 0.041)。他们更经常遭受其他后遗症(p -6)。大多数坏死涉及承重关节,69%的病例为多灶性坏死。对39名患者的核磁共振成像进行双盲回顾,发现14名患者出现严重病变,通常在髋关节。青少年和成人的生活质量较差,并在发病后受到永久性影响。结论:AL诊断时年龄>10岁、女性、复发率是多灶性ON的危险因素;MRI显示三分之一的患者有严重的ON;ON与影响多个领域的持续较差的生活质量有关。未来的研究应该包括关于ON管理的前瞻性数据,并寻求确定靶向筛查的遗传标记,从而实现ON的早期检测和治疗。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
CiteScore
2.60
自引率
5.90%
发文量
71
审稿时长
6-12 weeks
期刊介绍: PHO: Pediatric Hematology and Oncology covers all aspects of research and patient management within the area of blood disorders and malignant diseases of childhood. Our goal is to make PHO: Pediatric Hematology and Oncology the premier journal for the international community of clinicians and scientists who together aim to define optimal therapeutic strategies for children and young adults with cancer and blood disorders. The journal supports articles that address research in diverse clinical settings, exceptional case studies/series that add novel insights into pathogenesis and/or clinical care, and reviews highlighting discoveries and challenges emerging from consortia and conferences. Clinical studies as well as basic and translational research reports regarding cancer pathogenesis, genetics, molecular diagnostics, pharmacology, stem cells, molecular targeting, cellular and immune therapies and transplantation are of interest. Papers with a focus on supportive care, late effects and on related ethical, legal, psychological, social, cultural, or historical aspects of these fields are also appreciated. Reviews on important developments in the field are welcome. Articles from scientists and clinicians across the international community of Pediatric Hematology and Oncology are considered for publication. The journal is not dependent on or connected with any organization or society. All submissions undergo rigorous peer review prior to publication. Our Editorial Board includes experts in Pediatric Hematology and Oncology representing a wide range of academic and geographic diversity.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信