Giertz Mia, Aarnivala Henri, Wilk Michelsen Sascha, Björklund Caroline, Englund Annika, Grönroos Marika, Hjalgrim Lisa Lyngsie, Huttunen Pasi, Niinimäki Tuukka, Penno Eva, Pöyhönen Tuuli, Raittinen Päivi, Ranta Susanna, Svahn Johan E, Törnudd Lisa, Niinimäki Riitta, Harila Arja
Department of Women's and Children's Health, Uppsala University, Uppsala, Sweden.
Department of Pediatric Oncology and Hematology, Uppsala University Hospital, Uppsala, Sweden.
Pediatr Blood Cancer. 2024 Nov;71(11):e31250. doi: 10.1002/pbc.31250. Epub 2024 Aug 14.
Osteonecrosis (ON) is a potentially disabling skeletal complication of cancer treatment. Although symptomatic osteonecrosis (sON) is well-known in acute lymphoblastic leukemia (ALL), with an incidence around 6%, studies on sON in pediatric Hodgkin lymphoma (HL) are scarce. The aim of this study was to examine the incidence, risk factors, and outcome of sON in children treated for HL.
A total of 490 children under 18, diagnosed with HL between 2005 and 2019 in Sweden, Finland, and Denmark were eligible for the study. Data on patient characteristics, HL treatment, and development of sON were collected from patients' medical records. Magnetic resonance imaging scans were used to establish ON diagnosis and grade ON according to the Niinimäki grading system.
Cumulative 2-year incidence of sON among the 489 included patients was 5.5% (n = 30). The risk for developing sON was higher for those with older age (odds ratio [OR] 1.25, 95% confidence interval [CI]: 1.05-1.49, p < .010), female sex (OR 4.45, CI 1.87-10.58, p < .001), high total cumulative glucocorticoid (GC) doses (OR 1.76, 95% CI: 1.21-2.56, p = 0.003), and advanced HL (OR 2.19, 95% CI: 1.03-4.65, p = .042). Four (13.3%) patients underwent major surgical procedures and 13 (43.3%) had persistent symptoms due to ON at follow-up.
This study shows that sON is as common in pediatric HL as in pediatric ALL, with risk factors such as older age, female sex, high cumulative GC doses, and advanced HL. Future HL protocol development should aim to reduce the burden of ON by modifying GC treatment.
骨坏死（ON）是癌症治疗中一种可能导致残疾的骨骼并发症。虽然急性淋巴细胞白血病（ALL）中症状性骨坏死（sON）广为人知，发病率约为6%，但关于儿童霍奇金淋巴瘤（HL）中sON的研究却很少。本研究的目的是调查接受HL治疗的儿童中sON的发病率、危险因素及预后情况。
共有490名18岁以下儿童符合本研究条件，他们于2005年至2019年期间在瑞典、芬兰和丹麦被诊断为HL。从患者病历中收集了患者特征、HL治疗及sON发生情况的数据。采用磁共振成像扫描根据Niinimäki分级系统进行ON诊断并对ON进行分级。
489例纳入患者中sON的累积2年发病率为5.5%（n = 30）。年龄较大者（比值比[OR] 1.25，95%置信区间[CI]：1.05 - 1.49，p <.010）、女性（OR 4.45，CI 1.87 - 10.58，p <.001）、糖皮质激素（GC）总累积剂量高（OR 1.76，95% CI：1.21 - 2.56，p = 0.003）以及HL分期较晚（OR 2.19，95% CI：1.03 - 4.65，p =.042）的患者发生sON的风险更高。4例（13.3%）患者接受了大手术，13例（43.3%）患者在随访时因ON仍有持续症状。
本研究表明，sON在儿童HL中与在儿童ALL中一样常见，其危险因素包括年龄较大、女性、GC累积剂量高以及HL分期较晚。未来HL治疗方案的制定应旨在通过调整GC治疗来减轻ON的负担。
