Julia O'Mahony1, Ruth Ann Marrie2, Audrey Laporte3, E Ann Yeh4, Amit Bar-Or5, Cathy Phan6, David Buckley7, David Callen8, Mary B Connolly9, Daniela Pohl10, Marie-Emmanuelle Dilenge11, Geneviève Bernard11, Anne Lortie12, Noel Lowry13, E Athen MacDonald14, David Meek15, Guillaume Sébire11, Sunita Venkateswaran10, Ellen Wood16, Jerome Yager17, Brenda Banwell18. 1. Institute of Health Policy, Management, and Evaluation, University of Toronto, Toronto, Ontario, Canada; The Hospital for Sick Children, Toronto, Ontario, Canada; julia.omahony@mail.utoronto.ca. 2. Department of Internal Medicine, Winnipeg Health Sciences Center, University of Manitoba, Winnipeg, Manitoba, Canada; 3. Institute of Health Policy, Management, and Evaluation, University of Toronto, Toronto, Ontario, Canada; 4. Division of Neurology, The Hospital for Sick Children, Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada; 5. Neuroimmunology Unit, Montreal Neurologic Institute and Hospital, McGill University, Montreal, Quebec, Canada; Experimental Therapeutics Program, Montreal Neurologic Institute and Hospital, McGill University, Montreal, Quebec, Canada; 6. The Hospital for Sick Children, Toronto, Ontario, Canada; 7. Janeway Children's Health and Rehabilitation Centre, St John's, Newfoundland, Canada; 8. Department of Pediatrics, McMaster University, Hamilton, Ontario, Canada; 9. Department of Pediatrics, University of British Columbia, Vancouver, British Columbia, Canada; 10. Department of Pediatrics, Children's Hospital of Eastern Ontario, University of Ottawa, Ottawa, Ontario, Canada; 11. Department of Pediatrics, Montreal Children's Hospital, Montreal, Quebec, Canada; 12. Department of Pediatrics, Université de Montreal, Montreal, Quebec, Canada; 13. Department of Pediatrics, University of Saskatchewan, Saskatoon, Saskatchewan, Canada; 14. Department of Pediatrics, Queen's University, Kingston, Ontario, Canada; 15. Department of Pediatrics, Saint John Regional Hospital, Saint John, New Brunswick, Canada; 16. Department of Pediatrics, IWK Health Centre, Halifax, Nova Scotia, Canada; 17. Department of Pediatrics, Stollery Children's Hospital, Edmonton, Alberta, Canada; and. 18. The Hospital for Sick Children, Toronto, Ontario, Canada; The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania.
Abstract
BACKGROUND: Few prospective studies have systematically evaluated the extent of recovery from incident acquired demyelinating syndromes (ADS) of the central nervous system in children. METHODS: In a national cohort study of pediatric ADS, severity of the incident attack and extent of recovery by 12 months were evaluated. Annual evaluations were used to determine current diagnoses (monophasic ADS or multiple sclerosis [MS]) and new deficits. RESULTS: Of 283 children, 244 (86%) required hospitalization for a median (interquartile range [IQR]) of 6 (3-10) days, and 184 had moderate or severe deficits; 41 children were profoundly encephalopathic, 129 were unable to ambulate independently, and 59 with optic neuritis (ON) had moderately or severely impaired vision. Those with transverse myelitis (TM) and patients with monophasic disease were more likely to have moderate or severe deficits at onset. Twenty-seven children (10%) did not experience full neurologic recovery from their incident attack; 12 have severe residual deficits. Monophasic illness, TM, and moderate or severe deficits at onset were associated with poor recovery. After a median (IQR) follow-up of 5.06 (3.41-6.97) years, 59 children (21%) were diagnosed with MS; all recovered fully from their incident ADS attacks, although 6 subsequently acquired irreversible deficits after a median (IQR) observation period of 5.93 (4.01-7.02) years. CONCLUSIONS: ADS is a serious illness, with 86% of affected Canadian children requiring hospitalization. More than 90% of children recovered physically from their ADS event, including those children experiencing onset of MS. However, permanent visual or spinal cord impairment occurred in some children with ON or TM.
BACKGROUND: Few prospective studies have systematically evaluated the extent of recovery from incident acquired demyelinating syndromes (ADS) of the central nervous system in children. METHODS: In a national cohort study of pediatric ADS, severity of the incident attack and extent of recovery by 12 months were evaluated. Annual evaluations were used to determine current diagnoses (monophasic ADS or multiple sclerosis [MS]) and new deficits. RESULTS: Of 283 children, 244 (86%) required hospitalization for a median (interquartile range [IQR]) of 6 (3-10) days, and 184 had moderate or severe deficits; 41 children were profoundly encephalopathic, 129 were unable to ambulate independently, and 59 with optic neuritis (ON) had moderately or severely impaired vision. Those with transverse myelitis (TM) and patients with monophasic disease were more likely to have moderate or severe deficits at onset. Twenty-seven children (10%) did not experience full neurologic recovery from their incident attack; 12 have severe residual deficits. Monophasic illness, TM, and moderate or severe deficits at onset were associated with poor recovery. After a median (IQR) follow-up of 5.06 (3.41-6.97) years, 59 children (21%) were diagnosed with MS; all recovered fully from their incident ADS attacks, although 6 subsequently acquired irreversible deficits after a median (IQR) observation period of 5.93 (4.01-7.02) years. CONCLUSIONS: ADS is a serious illness, with 86% of affected Canadian children requiring hospitalization. More than 90% of children recovered physically from their ADS event, including those children experiencing onset of MS. However, permanent visual or spinal cord impairment occurred in some children with ON or TM.
Authors: Stacy L Pineles; Grant T Liu; Amy T Waldman; Elizabeth Lazar; Mark J Kupersmith; Michael X Repka Journal: J Neuroophthalmol Date: 2016-06 Impact factor: 3.042
Authors: Ruth Ann Marrie; Julia O'Mahony; Colleen J Maxwell; Vicki Ling; E Ann Yeh; Douglas L Arnold; Amit Bar-Or; Brenda Banwell Journal: PLoS One Date: 2019-06-11 Impact factor: 3.240
Authors: Giulia Fadda; Cesar A Alves; Julia O'Mahony; Denise A Castro; E Ann Yeh; Ruth Ann Marrie; Douglas L Arnold; Patrick Waters; Amit Bar-Or; Arastoo Vossough; Brenda Banwell Journal: JAMA Netw Open Date: 2021-10-01
Authors: Giulia Fadda; Patrick Waters; Mark Woodhall; Robert A Brown; Julia O'Mahony; Denise A Castro; Giulia Longoni; E Ann Yeh; Ruth Ann Marrie; Douglas L Arnold; Brenda Banwell; Amit Bar-Or Journal: Mult Scler Date: 2022-05-17 Impact factor: 5.855
Authors: Patrick Waters; Giulia Fadda; Mark Woodhall; Julia O'Mahony; Robert A Brown; Denise A Castro; Giulia Longoni; Sarosh R Irani; Bo Sun; E Ann Yeh; Ruth Ann Marrie; Douglas L Arnold; Brenda Banwell; Amit Bar-Or Journal: JAMA Neurol Date: 2020-01-01 Impact factor: 18.302