Children's Hospital and Pediatric Research Center, University of Helsinki, Helsinki University Hospital, Helsinki, Finland.
Research Program for Clinical and Molecular Metabolism, Faculty of Medicine, University of Helsinki, Helsinki, Finland.
Acta Oncol. 2021 Sep;60(9):1140-1145. doi: 10.1080/0284186X.2021.1950928. Epub 2021 Jul 26.
Osteonecrosis (ON) is a recognized complication of childhood ALL, but its optimal management remains unestablished. This study evaluated the effect of bisphosphonate (BP) treatment on the evolution of ON lesions in childhood ALL. We included a national cohort of ALL patients diagnosed with symptomatic ON before 18 years of age and treated with BPs ( = 10; five males). Patients were followed both clinically and with serial MRIs. ON lesions were graded according to the Niinimäki classification. The 10 patients had a total of 55 ON lesions. The median age was 13.3 years at ALL diagnosis and 14.8 years at ON diagnosis. Four patients had received HSCT before the ON diagnosis. BPs used were pamidronate ( = 7), alendronate ( = 2) and ibandronate ( = 1). The duration of BP treatment varied between 4 months and 4 years. In 4/10 patients, BP treatment was given during the chemotherapy. BPs were well-tolerated, with no severe complications or changes in kidney function. At the end of follow up 13/55 (24%) ON lesions were completely healed both clinically and radiographically; all these lesions were originally graded 3 or less. In contrast, ON lesions originally classified as grade 5 (joint destruction; = 4) remained at grade 5. All grade 5 hip joint lesions needed surgical treatment. During BP treatment, the pain was relieved in 7/10 patients. At the end of follow-up, none of the patients reported severe or frequent pain. BP treatment was safe and seemed effective in relieving ON-induced pain in childhood ALL. After articular collapse (grade 5) lesions did not improve with BP treatment. Randomized controlled studies are needed to further elucidate the role of BPs in childhood ALL-associated ON.
骨坏死（ON）是儿童 ALL 的一种公认并发症，但最佳治疗方法尚未确定。本研究评估了双膦酸盐（BP）治疗对儿童 ALL 中 ON 病变演变的影响。我们纳入了一个在 18 岁之前被诊断为症状性 ON 且接受 BP 治疗的 ALL 患儿的全国性队列（n=10；男 5 例）。患者同时接受临床和连续 MRI 随访。ON 病变根据 Niinimäki 分类进行分级。10 例患者共有 55 个 ON 病变。ALL 诊断时的中位年龄为 13.3 岁，ON 诊断时的中位年龄为 14.8 岁。4 例患者在 ON 诊断前接受过 HSCT。使用的 BP 为帕米膦酸盐（n=7）、阿仑膦酸盐（n=2）和伊班膦酸盐（n=1）。BP 治疗时间在 4 个月至 4 年之间不等。在 10 例患者中的 4 例中，在化疗期间给予 BP 治疗。BP 治疗耐受性良好，无严重并发症或肾功能变化。在随访结束时，13/55（24%）的 ON 病变在临床和影像学上均完全愈合；所有这些病变最初的分级均为 3 级或更低。相比之下，最初分级为 5 级（关节破坏；n=4）的 ON 病变仍保持在 5 级。所有 5 级髋关节病变均需要手术治疗。在 BP 治疗期间，10 例患者中有 7 例疼痛缓解。在随访结束时，没有患者报告严重或频繁疼痛。BP 治疗安全且似乎有效，可缓解儿童 ALL 相关 ON 引起的疼痛。在关节塌陷（5 级）后，BP 治疗不能改善病变。需要进行随机对照研究以进一步阐明 BP 在儿童 ALL 相关 ON 中的作用。
