Kittivisuit Sirinthip, Sripornsawan Pornpun, Songthawee Natsaruth, Chavananon Shevachut, McNeil Edward B, Chotsampancharoen Thirachit
Department of Pediatrics, Faculty of Medicine, Prince of Songkla University, Hat Yai, Thailand.
Epidemiology Unit, Faculty of Medicine, Prince of Songkla University, Hat Yai, Thailand.
Pediatr Rheumatol Online J. 2022 May 2;20(1):34. doi: 10.1186/s12969-022-00692-9.
BACKGROUND : Childhood leukemia with musculoskeletal (MSK) involvement mimics various conditions, which consequently leads to diagnostic delays. The clinical implication of MSK involvement in this disease on survival outcomes is inconclusive. This study aimed to compare characteristics and survival outcomes between MSK and non-MSK involvement in childhood leukemia.
The medical records of children newly diagnosed with acute leukemia of an age under 15 years were retrospectively reviewed. Two-to-one nearest-neighbor propensity score-matching was performed to obtain matched groups with and without MSK involvement. The Kaplan-Meier method and log-rank test were then used to assess the effect of MSK involvement on survival outcomes.
Of 1042 childhood leukemia cases, 81 (7.8%) children had MSK involvement at initial presentation. MSK involvement was more likely in children with acute lymphoblastic leukemia than acute myeloid leukemia (p < 0.05). Hematologic abnormalities were less frequent in the MSK involvement group (p < 0.05). The absence of peripheral blast cells was significantly higher in the MSK involvement group (17.3% vs 9.6%, p = 0.04). Normal complete blood counts with absence of peripheral blast cells were found 2.5% of the children with MSK involvement. By propensity score-matching for comparable risk groups of children with and without MSK involvement, the 5-year overall survival was not significantly different (48.2% vs 57.4%, respectively, p = 0.22), nor was event-free survival (43.3% vs 51.8%, respectively, p = 0.31).
Childhood leukemia with MSK involvement had the characteristics of minimal or absent hematologic abnormalities and peripheral blast counts.
背景:伴有肌肉骨骼(MSK)受累的儿童白血病可模拟多种病症,从而导致诊断延迟。MSK受累在该疾病中对生存结局的临床影响尚无定论。本研究旨在比较儿童白血病中MSK受累与非MSK受累的特征及生存结局。
回顾性分析15岁以下新诊断为急性白血病儿童的病历。采用二比一最近邻倾向得分匹配法,以获得有和无MSK受累的匹配组。然后使用Kaplan-Meier法和对数秩检验来评估MSK受累对生存结局的影响。
在1042例儿童白血病病例中,81例(7.8%)儿童在初次就诊时有MSK受累。急性淋巴细胞白血病儿童比急性髓细胞白血病儿童更易发生MSK受累(p<0.05)。MSK受累组血液学异常的发生率较低(p<0.05)。MSK受累组外周血原始细胞缺失率显著更高(17.3%对9.6%,p=0.04)。在有MSK受累的儿童中,2.5%的儿童全血细胞计数正常且外周血无原始细胞。通过对有和无MSK受累的可比风险儿童组进行倾向得分匹配,5年总生存率无显著差异(分别为48.2%对57.4%,p=0.22),无事件生存率也无显著差异(分别为43.3%对51.8%,p=0.31)。
伴有MSK受累的儿童白血病具有血液学异常轻微或无异常以及外周血原始细胞计数少的特征。