Line Kenborg1, Jeanette Falck Winther1, Karen Markussen Linnet2, Anja Krøyer1, Vanna Albieri3, Anna Sällfors Holmqvist4, Laufey Tryggvadottir5,6, Laura Maria Madanat-Harjuoja7, Marilyn Stovall8, Henrik Hasle2, Jørgen H Olsen9. 1. Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark. 2. Department of Pediatrics, Aarhus University Hospital, Aarhus, Denmark. 3. Unit of Statistics and Pharmacoepidemiology, Danish Cancer Society Research Center, Copenhagen, Denmark. 4. Department of Clinical Sciences, Lund University, Pediatric Oncology and Hematology, Skåne University Hospital, Lund, Sweden. 5. The Icelandic Cancer Registry, Reykjavik, Iceland. 6. Faculty of Medicine, University of Iceland, Reykjavik, Iceland. 7. The Finnish Cancer Registry, Helsinki, Finland. 8. Department of Radiation Physics, The University of Texas MD Anderson Cancer Center, Texas, Houston, USA. 9. Research Management and Administration, Danish Cancer Society Research Center, Copenhagen, Denmark.
Abstract
Background: A comprehensive overview of neurologic complications among survivors of central nervous system (CNS) tumors in childhood is lacking. We aimed to investigate the risk for these disorders in a large, population-based study with outcome measures from nationwide hospital registries. Methods: We identified 4858 five-year survivors with diagnoses of CNS tumor in childhood in Denmark, Iceland, Finland, and Sweden in 1943-2007, and 166658 matched population comparison subjects. Inpatient discharge diagnoses of neurologic disorders were used to calculate relative risks (RRs) and absolute excess risks (AERs). Results: A neurologic disorder was verified in 1309 survivors, while 92.4 were expected, yielding an overall RR of 14.2 (95% confidence interval [CI]: 13.3-15.1) and an AER of 20 hospitalizations per 1000 persons per year. The risks remained increased more than 20 years after diagnosis (RR: 6.3, 95% CI: 5.6-7.2; AER: 11, 9-12). The most frequent diagnoses were epilepsy (affecting 14.1% of all survivors) followed by hydrocephalus (9.5%) and paralytic syndromes (4.2%), with RRs of 28.7 (95% CI: 26.0-31.6), 243 (95% CI: 190-311), and 40.3 (95% CI: 33.1-49.2), respectively. Of these outcomes, 30%-40% were diagnosed prior to or synchronously with the CNS tumor. The survivors had highly increased RRs for infectious diseases of the CNS, disorders of cranial nerves, and degenerative diseases of the nervous system. Conclusions: Survivors of childhood CNS tumors are at markedly increased risk for neurologic disorders throughout their lives. Health care professionals must be aware of survivors who might benefit from preventive interventions and intensive follow-up.
Background: A comprehensive overview of neurologic complications among survivors of central nervous system (CNS) tumors in childhood is lacking. We aimed to investigate the risk for these disorders in a large, population-based study with outcome measures from nationwide hospital registries. Methods: We identified 4858 five-year survivors with diagnoses of CNS tumor in childhood in Denmark, Iceland, Finland, and Sweden in 1943-2007, and 166658 matched population comparison subjects. Inpatient discharge diagnoses of neurologic disorders were used to calculate relative risks (RRs) and absolute excess risks (AERs). Results:A neurologic disorder was verified in 1309 survivors, while 92.4 were expected, yielding an overall RR of 14.2 (95% confidence interval [CI]: 13.3-15.1) and an AER of 20 hospitalizations per 1000 persons per year. The risks remained increased more than 20 years after diagnosis (RR: 6.3, 95% CI: 5.6-7.2; AER: 11, 9-12). The most frequent diagnoses were epilepsy (affecting 14.1% of all survivors) followed by hydrocephalus (9.5%) and paralytic syndromes (4.2%), with RRs of 28.7 (95% CI: 26.0-31.6), 243 (95% CI: 190-311), and 40.3 (95% CI: 33.1-49.2), respectively. Of these outcomes, 30%-40% were diagnosed prior to or synchronously with the CNS tumor. The survivors had highly increased RRs for infectious diseases of the CNS, disorders of cranial nerves, and degenerative diseases of the nervous system. Conclusions: Survivors of childhood CNS tumors are at markedly increased risk for neurologic disorders throughout their lives. Health care professionals must be aware of survivors who might benefit from preventive interventions and intensive follow-up.
Authors: Bjørn Møller; Harald Fekjaer; Timo Hakulinen; Laufey Tryggvadóttir; Hans H Storm; Mats Talbäck; Tor Haldorsen Journal: Eur J Cancer Prev Date: 2002-06 Impact factor: 2.497
Authors: Daniel M Green; Larry E Kun; Katherine K Matthay; Anna T Meadows; William H Meyer; Paul A Meyers; Sheri L Spunt; Leslie L Robison; Melissa M Hudson Journal: Pediatr Blood Cancer Date: 2013-02-15 Impact factor: 3.167
Authors: Stanislaw Garwicz; Harald Anderson; Jørgen H Olsen; Jeanette Falck Winther; Risto Sankila; Frøydis Langmark; Laufey Tryggvadóttir; Torgil R Möller Journal: Int J Cancer Date: 2012-01-11 Impact factor: 7.396
Authors: Gregory T Armstrong; Wilburn E Reddick; Ronald C Petersen; Aimee Santucci; Nan Zhang; Deokumar Srivastava; Robert J Ogg; Claudia M Hillenbrand; Noah Sabin; Matthew J Krasin; Larry Kun; Ching-Hon Pui; Melissa M Hudson; Leslie L Robison; Kevin R Krull Journal: J Natl Cancer Inst Date: 2013-04-12 Impact factor: 13.506
Authors: Thorgerdur Gudmundsdottir; Jeanette F Winther; Sofie de Fine Licht; Trine G Bonnesen; Peter H Asdahl; Laufey Tryggvadottir; Harald Anderson; Finn Wesenberg; Nea Malila; Henrik Hasle; Jørgen H Olsen Journal: Int J Cancer Date: 2015-03-14 Impact factor: 7.396
Authors: Elizabeth M Wells; Nicole J Ullrich; Kristy Seidel; Wendy Leisenring; Charles A Sklar; Gregory T Armstrong; Lisa Diller; Allison King; Kevin R Krull; Joseph P Neglia; Marilyn Stovall; Kimberly Whelan; Kevin C Oeffinger; Leslie L Robison; Roger J Packer Journal: Neuro Oncol Date: 2018-01-10 Impact factor: 12.300
Authors: Roger J Packer; James G Gurney; Judy A Punyko; Sarah S Donaldson; Peter D Inskip; Marilyn Stovall; Yutaka Yasui; Ann C Mertens; Charles A Sklar; H Stacy Nicholson; Lonnie K Zeltzer; Joseph P Neglia; Leslie L Robison Journal: J Clin Oncol Date: 2003-09-01 Impact factor: 50.717
Authors: Elizabeth Schepke; Maja Löfgren; Torsten Pietsch; Thomas Olsson Bontell; Teresia Kling; Anna Wenger; Sandra Ferreyra Vega; Anna Danielsson; Sandor Dosa; Stefan Holm; Anders Öberg; Per Nyman; Marie Eliasson-Hofvander; Per-Erik Sandström; Stefan M Pfister; Birgitta Lannering; Magnus Sabel; Helena Carén Journal: Neuropathol Appl Neurobiol Date: 2022-08-03 Impact factor: 6.250
Authors: Anne Sophie Lind Helligsoe; Kathrine Synne Weile; Line Kenborg; Louise Tram Henriksen; Yasmin Lassen-Ramshad; Ali Amidi; Lisa Maria Wu; Jeanette Falck Winther; Line Pickering; René Mathiasen Journal: Front Neurosci Date: 2022-02-22 Impact factor: 4.677