Stavroula Anastasopoulou1, Mats A Eriksson1, Mats Heyman1, Chen Wang1, Riitta Niinimäki2, Sirje Mikkel3, Goda E Vaitkevičienė4, Inga Maria Johannsdottir5, Ida Hed Myrberg6, Olafur Gisli Jonsson7, Bodil Als-Nielsen8, Kjeld Schmiegelow8, Joanna Banerjee9, Arja Harila-Saari10, Susanna Ranta1. 1. Department of Women's and Children's Health, Karolinska Institute and Karolinska University Hospital, Stockholm, Sweden. 2. Oulu University Hospital, Department of Children and Adolescents, and University of Oulu, PEDEGO Research Unit, Oulu, Finland. 3. Department of Hematology and Oncology, University of Tartu, Tartu, Estonia. 4. Children's Hospital, affiliation of Vilnius University Hospital Santaros Klinikos and Vilnius University, Vilnius, Lithuania. 5. Department of Pediatric Hematology/Oncology, Oslo University Hospital, Oslo, Norway. 6. Department of Women's and Children's Health, Karolinska Institute, Stockholm, Sweden. 7. Department of Pediatrics, University of Iceland, Reykjavík, Iceland. 8. Department of Pediatrics and Adolescent Medicine, University Hospital Rigshospitalet, Copenhagen, and Institute of Clinical Medicine, Faculty of Medicine, University of Copenhagen, Denmark. 9. Department of Pediatric Hematology-Oncology and Stem Cell Transplantation, Children's Hospital, University of Helsinki and Helsinki University Hospital, Helsinki, Finland. 10. Department of Women's and Children's Health, University of Uppsala, Uppsala, Sweden.
Abstract
BACKGROUND: Posterior reversible encephalopathy syndrome (PRES) is a distinct entity with incompletely known predisposing factors. The aim of this study is to describe the incidence, risk factors, clinical course, and outcome of PRES in childhood acute lymphoblastic leukemia (ALL). PROCEDURE: Patients aged 1.0 to 17.9 years diagnosed with ALL from July 2008 to December 2015 and treated according to the Nordic Society of Pediatric Hematology and Oncology (NOPHO) ALL2008 protocol were included. Patients with PRES were identified in the prospective NOPHO leukemia toxicity registry, and clinical data were collected from the medical records. RESULTS: The study group included 1378 patients, of whom 52 met the criteria for PRES. The cumulative incidence of PRES at one month was 1.7% (95% CI, 1.1-2.5) and at one year 3.7% (95% CI, 2.9-4.9). Older age (hazard ratios [HR] for each one-year increase in age 1.1; 95% CI, 1.0-1.2, P = 0.001) and T-cell immunophenotype (HR, 2.9; 95% CI, 1.6-5.3, P = 0.0005) were associated with PRES. Central nervous system (CNS) involvement (odds ratios [OR] = 2.8; 95% CI, 1.2-6.5, P = 0.015) was associated with early PRES and high-risk block treatment (HR = 2.63; 95% CI, 1.1-6.4, P = 0.033) with late PRES. At follow-up of the PRES patients, seven patients had epilepsy and seven had neurocognitive difficulties. CONCLUSION: PRES is a neurotoxicity in the treatment of childhood ALL with both acute and long-term morbidity. Older age, T-cell leukemia, CNS involvement and high-risk block treatment are risk factors for PRES.
BACKGROUND: Posterior reversible encephalopathy syndrome (PRES) is a distinct entity with incompletely known predisposing factors. The aim of this study is to describe the incidence, risk factors, clinical course, and outcome of PRES in childhood acute lymphoblastic leukemia (ALL). PROCEDURE: Patients aged 1.0 to 17.9 years diagnosed with ALL from July 2008 to December 2015 and treated according to the Nordic Society of Pediatric Hematology and Oncology (NOPHO) ALL2008 protocol were included. Patients with PRES were identified in the prospective NOPHO leukemia toxicity registry, and clinical data were collected from the medical records. RESULTS: The study group included 1378 patients, of whom 52 met the criteria for PRES. The cumulative incidence of PRES at one month was 1.7% (95% CI, 1.1-2.5) and at one year 3.7% (95% CI, 2.9-4.9). Older age (hazard ratios [HR] for each one-year increase in age 1.1; 95% CI, 1.0-1.2, P = 0.001) and T-cell immunophenotype (HR, 2.9; 95% CI, 1.6-5.3, P = 0.0005) were associated with PRES. Central nervous system (CNS) involvement (odds ratios [OR] = 2.8; 95% CI, 1.2-6.5, P = 0.015) was associated with early PRES and high-risk block treatment (HR = 2.63; 95% CI, 1.1-6.4, P = 0.033) with late PRES. At follow-up of the PRES patients, seven patients had epilepsy and seven had neurocognitive difficulties. CONCLUSION: PRES is a neurotoxicity in the treatment of childhood ALL with both acute and long-term morbidity. Older age, T-cell leukemia, CNS involvement and high-risk block treatment are risk factors for PRES.
Authors: Judit C Sági; András Gézsi; Bálint Egyed; Zsuzsanna Jakab; Noémi Benedek; Andishe Attarbaschi; Stefan Köhrer; Jakub Sipek; Lucie Winkowska; Marketa Zaliova; Stavroula Anastasopoulou; Benjamin Ole Wolthers; Susanna Ranta; Csaba Szalai; Gábor T Kovács; Ágnes F Semsei; Dániel J Erdélyi Journal: Cancers (Basel) Date: 2021-05-12 Impact factor: 6.639