Recovery From Central Nervous System Acute Demyelination in Children

O’Mahony, Julia; Marrie, Ruth Ann; Laporte, Audrey; Yeh, E. Ann; Bar‐Or, Amit; Phan, Cathy; Buckley, David; Callen, David; Connolly, Mary; Pohl, Daniela; Dilenge, Marie-Emmanuelle; Bernard, Geneviève; Lortie, Anne; Lowry, Noel J.; Macdonald, Eilidh; Meek, David; Sébire, Guillaume; Venkateswaran, Sunita; Wood, Ellen; Yager, Jerome Y.; Banwell, Brenda

doi:10.1542/peds.2015-0028

Cited by 44 publications

(40 citation statements)

References 25 publications

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“…For example, in one study that followed 59 patients with multiple sclerosis for a median of 5 years 11 months, 90% continued to have a normal neurological examination at final follow-up. 33 In a German study of 88 children with multiple sclerosis, the median scores on the Expanded Disability Status Scale were less than 1 at 2 years, 1.2 at 10 years, and 2.5 at 15 years. 29 A seminal paper that compared 394 patients with paediatric-onset multiple sclerosis and 1775 patients with adult-onset multiple sclerosis demonstrated that it took approximately 10 years longer for the patients with paediatric-onset disease to reach irreversible disability and secondary progression; however, they reached these landmarks at a biological age approximately 10 years younger than their counterparts with the adult-onset disease.…”

Section: Clinical Features and Disease Course: Does The Age Matter?mentioning

confidence: 96%

“…This is postulated to be due to the greater plasticity of the developing brain and consequently a greater capacity for repair. For example, in one study that followed 59 patients with multiple sclerosis for a median of 5 years 11 months, 90% continued to have a normal neurological examination at final follow‐up . In a German study of 88 children with multiple sclerosis, the median scores on the Expanded Disability Status Scale were less than 1 at 2 years, 1.2 at 10 years, and 2.5 at 15 years .…”

Section: Clinical Features and Disease Course: Does The Age Matter?mentioning

confidence: 99%

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Paediatric multiple sclerosis: a new era in diagnosis and treatment

Duignan

Brownlee

Wassmer

et al. 2019

Develop Med Child Neuro

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Multiple sclerosis is a chronic immune‐mediated demyelinating disease of the central nervous system. The diagnosis of multiple sclerosis in children, as in adults, requires evidence of dissemination of inflammatory activity in more than one location in the central nervous system (dissemination in space) and recurrent disease over time (dissemination in time). The identification of myelin oligodendrocyte glycoprotein antibodies (MOG‐Ab) and aquaporin‐A antibodies (AQP4‐Ab), and the subsequent discovery of their pathogenic mechanisms, have led to a shift in the classification of relapsing demyelinating syndromes. This is reflected in the 2017 revised criteria for the diagnosis of multiple sclerosis, which emphasizes the exclusion of multiple sclerosis mimics and aims to enable earlier diagnosis and thus treatment initiation. The long‐term efficacy of individual therapies initiated in children with multiple sclerosis is hard to evaluate, owing to the small numbers of patients who have the disease, the relatively high number of patients who switch therapy, and the need for long follow‐up studies. Nevertheless, an improvement in prognosis with a globally reduced annual relapse rate in children with multiple sclerosis is now observed compared with the pretreatment era, indicating a possible long‐term effect of therapies. Given the higher relapse rate in children compared with adults, and the impact multiple sclerosis has on cognition in the developing brain, there is a question whether rapid escalation or potent agents should be used in children, while the short‐ and long‐term safety profiles of these drugs are being established. With the results of the first randomized controlled trial of fingolimod versus interferon‐β1a in paediatric multiple sclerosis published in 2018 and several clinical trials underway, there is hope for further progress in the field of paediatric multiple sclerosis. What this paper adds Early and accurate diagnosis of multiple sclerosis is crucial. The discovery of antibody‐mediated demyelination has changed the diagnosis and management of relapsing demyelination syndromes. Traditional escalation therapy is being challenged by induction therapy.

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Section: Clinical Features and Disease Course: Does The Age Matter?mentioning

confidence: 96%

Section: Clinical Features and Disease Course: Does The Age Matter?mentioning

confidence: 99%

Paediatric multiple sclerosis: a new era in diagnosis and treatment

Duignan

Brownlee

Wassmer

et al. 2019

Develop Med Child Neuro

View full text Add to dashboard Cite

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“…21 Three additional studies have also shown complete recovery after a first attack of MS. Of 236 pediatric patients with MS from the United Kingdom and Iraq included in these 3 studies, 176 (75%) recovered fully after the first event. 6,10,22 African ancestry is often associated with increased MS severity in North American adult patients with MS.…”

Section: E1-e18mentioning

confidence: 97%

Pediatric multiple sclerosis

et al. 2016

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Multiple sclerosis (MS) in children manifests with a relapsing-remitting MS (RRMS) disease course. Acute relapses consist of new neurologic deficits persisting greater than 24 hours, in the absence of intercurrent illness, and occur with a higher frequency early in the disease as compared to adult-onset RRMS. Most pediatric patients with MS recover well from these early relapses, and cumulative physical disability is rare in the first 10 years of disease. Brainstem attacks, poor recovery from a single attack, and a higher frequency of attacks portend a greater likelihood of future disability. Although prospective pediatric-onset MS cohorts have been established in recent years, there remains very limited prospective data detailing the longer-term clinical outcome of pediatric-onset MS into adulthood. Whether the advent of MS therapies, and the largely off-label access to such therapies in pediatric MS, has improved prognosis is unknown. MS onset during the key formative academic years, concurrent with active cognitive maturation, is an important determinant of long-term outcome, and is discussed in detail in another article in this supplement. Finally, increasing recognition of pediatric MS worldwide, recent launch of phase III trials for new agents in the pediatric MS population, and the clear imperative to more fully appreciate health-related quality of life in pediatric MS through adulthood highlight the need for standardized, validated, and robust outcome measures. Neurology ® 2016;87 (Suppl 2):S74-S81

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“…22 The tendency for very young patients with MS to manifest with polyfocal neurologic deficits and a high frequency of brainstem symptoms at onset were noted. Over the next 5 years, several key observations emerged: (1) children and adolescents experience relapsing neurologic deficits typical of adult-onset relapsingremitting MS but do not appear to experience primary progressive MS 23 ; (2) CSF analysis reveals the presence of intrathecal oligoclonal bands, a hallmark of MS in adults, in 60%-95% of pediatric patients, but less commonly in younger children 24 ; (3) over 95% of pediatric patients with MS recover well, in terms of physical functioning, from acute episodes early in their disease 25 ; (4) cognitive impairment occurs in approximately 30% of pediatric patients with MS [26][27][28][29][30][31][32] and correlates with MRI measures (reduced brain volume, altered resting state and functional connectivity, and reduced tissue integrity) [33][34][35][36][37][38] ; (5) pediatric patients with MS have higher relapse frequency in the first few years postonset, as compared to patients with adult-onset MS 39,40 ; and (6) time from first attack to secondary progressive MS (a stage of the disease characterized by progressive accrual of neurologic impairment not linked to discrete relapses) appears to take approximately 10 years longer in pediatric-onset patients, although the age at which such deficits appear occurs at a younger age than occurs in adult-onset MS.…”

Section: Pediatric Multiple Sclerosismentioning

confidence: 99%

Pediatric multiple sclerosis

Banwell

2016

Neurology

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In 1952, Sydney Carter, MD (1912MD ( -2005; Professor Emeritus, Columbia University), was asked to establish the first independent child neurology program at Columbia Presbyterian Hospital. Prior to this, children with neurologic disorders were largely cared for by general pediatricians, or on occasion, and often with trepidation, by adult neurologists. Dr. Carter recognized the unique care needs of the pediatric neurology patient. He prioritized the requirement to harmonize neuroscience and neurodevelopment, and advocated the need to identify the hallmarks of neurologic illness in the context of the developing central and peripheral nervous systems, and to foster training programs specifically in Child Neurology.It is my sincere honor to be the 2015 Sydney Carter Award recipient, and to pay service to the many colleagues whose dedication, brilliance, and compassion have contributed immeasurably to the content of this lecture. In my lecture, and herein, I summarize the learning curve and milestones achieved in pediatric multiple sclerosis (MS) care and research to date. I have endeavored to mention the work of international colleagues and to emphasize the Canadian Pediatric Demyelinating Disease program. The Canadian program began with a 3-year survey of over 2,400 pediatric health care providers 1-3 to estimate the annual incidence of acute demyelination in Canadian children. 4 Through the creation of the Canadian Pediatric Demyelinating Disease Network with funding by the Multiple Sclerosis Scientific Research Foundation, a prospective cohort (2004-present), a centralized database, biorepository, and high-quality research MRI dataset have contributed to our understanding of the clinical, genetic, epidemiologic, and imaging characteristics of MS in children.Just as Dr. Carter identified a fundamental need for pediatric neurology, formal programs for pediatriconset MS were also born from recognition of a clinical care need. Before 1980, search of the English literature yielded fewer than 30 publications on the topic of pediatric MS, and many believed that MS was a disease solely of adults (reviewed in Ref. 5). The available MS diagnostic criteria proposed by Poser et al.6 specifically excluded the diagnosis of MS in persons younger than 10 years, and did not formally comment on MS in youth. Further complicating the diagnosis and care of pediatric patients with MS is the fact that approximately 70% of pediatric patients with acute demyelination will have a transient illness without clinical or MRI evidence of new lesions over time. Acute disseminated encephalomyelitis (ADEM) typifies monophasic demyelination, particularly in young children. Distinguishing such patients from the 30% ultimately confirmed to have MS has been the focus of much of the research performed in the last 15 years. 7-9The diagnosis of MS rests on clinical features. There are no diagnostically specific genetic mutations or biomarkers for MS. Given this, an initial priority was to define the clinical features of pediatric-onset MS. Since 1980...

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Recovery From Central Nervous System Acute Demyelination in Children

Abstract: Few prospective studies have systematically evaluated the extent of recovery from incident acquired demyelinating syndromes (ADS) of the central nervous system in children.

Cited by 44 publications

References 25 publications

Paediatric multiple sclerosis: a new era in diagnosis and treatment

Paediatric multiple sclerosis: a new era in diagnosis and treatment

Pediatric multiple sclerosis

Pediatric multiple sclerosis

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