A1 Refereed original research article in a scientific journal
Symptomatic osteonecrosis in children treated for Hodgkin lymphoma: A population-based study in Sweden, Finland, and Denmark
Authors: Giertz, Mia; Aarnivala, Henri; Michelsen, Sascha Wilk; 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
Publisher: WILEY
Publishing place: HOBOKEN
Publication year: 2024
Journal: Pediatric Blood and Cancer
Journal name in source: PEDIATRIC BLOOD & CANCER
Journal acronym: PEDIATR BLOOD CANCER
Article number: e31250
Volume: 17
Issue: 11
Number of pages: 11
ISSN: 1545-5009
eISSN: 1545-5017
DOI: https://doi.org/10.1002/pbc.31250
Web address : https://doi.org/10.1002/pbc.31250
Self-archived copy’s web address: https://research.utu.fi/converis/portal/detail/Publication/457653434
Background: 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.
Procedure: 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 Niinim & auml;ki grading system.
Results: 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.
Conclusions: 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.
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Funding information in the publication:
We would like to thank Olle Lindinger, Umeå University Hospital (Sweden), Yvonne Håkansson, Lund University Hospital (Sweden), and Lars Kawan, Queen Silvia's Hospital in Gothenburg (Sweden), Steen Roshoej, Margrethe Ottosen Møller, and Hilde Dragland Galsgaard Aalborg, Aarhus and Odense (Denmark), for assistance in collecting data for the study. This study was supported by grants from theMary Beve's Foundation, Gerd Ahlman's Fund, Swedish Childhood Cancer Fund, Alma and K. A. Snellman Foundation, and the Väre Foundation for Pediatric Cancer Research, Danish Childhood Cancer Foundation (no. 2021-7439), Lion's Cancer Research Fund of Middle Sweden.