Patients with paediatric-onset multiple sclerosis are at higher risk of cognitive impairment in adulthood: An Italian collaborative study

Ruano, Luís; Branco, Mariana; Portaccio, Emilio; Goretti, Benedetta; Niccolai, Claudia; Patti, Francesco; Chisari, Clara Grazia; Gallo, Paolo; Grossi, Paola; Ghezzi, Angelo; Roscio, Marco; Mattioli, Flavia; Stampatori, Chiara; Simone, Marta; Viterbo, Rosa Gemma; Amato, Maria Pia

doi:10.1177/1352458517717341

Cited by 38 publications

(32 citation statements)

References 33 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…Cognitive impairment in childhood (Amato et al, 2016;Julian et al, 2013) and later on in adulthood (Ruano et al, 2018) in these patients may be the result of disturbed neural communication due to white and grey matter injury (Arrondo et al, 2009). In that regard, disrupted gamma oscillations are observed in adults with MS (Arpin et al, 2017;Barratt et al, 2017;Stickland et al, 2018).…”

Section: Use Of Inflammatory Demyelination As a Model To Examine Thmentioning

confidence: 99%

White matter plasticity and maturation in human cognition

Bells

Lefebvre

Longoni

et al. 2019

Glia

View full text Add to dashboard Cite

White matter plasticity likely plays a critical role in supporting cognitive development. However, few studies have used the imaging methods specific to white matter tissue structure or experimental designs sensitive to change in white matter necessary to elucidate these relations. Here we briefly review novel imaging approaches that provide more specific information regarding white matter microstructure. Furthermore, we highlight recent studies that provide greater clarity regarding the relations between changes in white matter and cognition maturation in both healthy children and adolescents and those with white matter insult. Finally, we examine the hypothesis that white matter is linked to cognitive function via its impact on neural synchronization. We test this hypothesis in a population of children and adolescents with recurrent demyelinating syndromes. Specifically, we evaluate group differences in white matter microstructure within the optic radiation; and neural phase synchrony in visual cortex during a visual task between 25 patients and 28 typically developing age‐matched controls. Children and adolescents with demyelinating syndromes show evidence of myelin and axonal compromise and this compromise predicts reduced phase synchrony during a visual task compared to typically developing controls. We investigate one plausible mechanism at play in this relationship using a computational model of gamma generation in early visual cortical areas. Overall, our findings show a fundamental connection between white matter microstructure and neural synchronization that may be critical for cognitive processing. In the future, longitudinal or interventional studies can build upon our knowledge of these exciting relations between white matter, neural communication, and cognition.

show abstract

Section: Use Of Inflammatory Demyelination As a Model To Examine Thmentioning

confidence: 99%

White matter plasticity and maturation in human cognition

Bells

Lefebvre

Longoni

et al. 2019

Glia

View full text Add to dashboard Cite

show abstract

“…In contrast, the highest ARR in Europe was reported to be 1.1 in patients ≤11 years old (37). Emerging evidence suggests that relapse rates may be higher among patients with pediatric MS than those with adultonset MS (30,39,95). In one study, relapse rates were also noted to be higher among children younger than 11 years old than among those 14-16 years old (37).…”

Section: Additional Files 3-6)mentioning

confidence: 95%

“…Three studies compared relapse rates amongst pediatric and adult patients with MS, finding that ARRs were consistently higher among pediatric patients (30,38,39). As expected, for both the pediatric and the adult population, ARRs were higher before treatment (2.76; 1.78) than after treatment (1.12; 0.35).…”

Section: Relapse Ratesmentioning

confidence: 99%

“…Ten studies reported fatigue-related outcomes among European patients with pediatric MS (see Additional File 4). Nine of these publications reported categorical outcomes, most of which assessed the percentage of patients experiencing fatigue, which ranged from 20% (60,61) to as high as 79.5% (38). Two publications reported continuous outcomes using either the Checklist Individual Strength fatigue score or the PedsQL MFS.…”

Section: Fatiguementioning

confidence: 99%

“…One study reported that the cognitive impairment associated with pediatric MS deteriorates over time (83). Two studies included patients with adult MS as a comparator; one of these studies found that patients with pediatric-onset MS are at a higher risk of developing cognitive impairment in adulthood than patients with adult-onset MS (38). Across studies, the percentage of patients with pediatric MS and cognitive impairment ranged between 18% and 70% (37,(85)(86)(87)(88)(89).…”

Section: Ten Studies Reported Cognitive Function Outcomes Among Europmentioning

confidence: 99%

See 2 more Smart Citations

A systematic literature review of the burden of pediatric multiple sclerosis in North America and Europe

Lb¹,

Goble²,

Kroes³

et al. 2019

Preprint

View full text Add to dashboard Cite

Background: Multiple sclerosis (MS) is a chronic neurodegenerative condition that severely impacts patient functioning and quality of life (QoL). Onset of MS typically occurs in adults and evidence is growing for onset in children and adolescents. Several studies describe the clinical burden of pediatric MS; however, to date, no systematic literature review summarizing the clinical burden of pediatric MS has been published. Objective: To conduct a systematic literature review to identify published data on the epidemiology, disease characteristics, QoL, and economic burden of pediatric MS in North America and Europe. Methods: Systematic searches were conducted in Ovid MEDLINE®, Embase, and Cochrane library to identify studies that enrolled patients with pediatric MS and reported at least one outcome related to the epidemiological, humanistic, and economic burden of pediatric MS. Only English-language studies were included. Results: In total, 7,447 unique records were identified, of which 103 met the study’s inclusion criteria. Of the included articles, 9 reported incidence and prevalence, 39 reported relapse rates, 15 reported QoL, 22 reported fatigue, 37 reported cognitive function, and 17 reported school performance. Incidence of pediatric MS ranged from 0.30 to 2.64 per 100,000 person-years, with few studies suggesting that incidence may increase with age. Prevalence was reported as high as 14.6 cases per 100,000 persons in the USA and 9.6% of all MS patients in European countries. The range of annualized relapse rates was similar across countries, at 0.36-2.76 in North America and 0.65-1.11 in European countries. Patients with pediatric MS experienced reduced QoL compared to healthy controls. Patients experienced reduced diminished cognitive functioning, worse school performance and increased fatigue. Limited information was identified for the mortality and economic burden of pediatric MS. Conclusions: Several studies describe the clinical burden of pediatric MS. Although incidence and prevalence rates for the disease are relatively low, the rate of relapse and burden to patients is considerable. Further studies are required to address gaps in the understanding of prognostic factors for relapse and the impact of QoL, fatigue, and cognitive function. Additional research on mortality and economic outcomes will be important to guide treatment pathways and reimbursement decisions.

show abstract

Long-term Cognitive Consequences for Patients With Pediatric-Onset Multiple Sclerosis

Krupp

Charvet

2019

JAMA Neurol

View full text Add to dashboard Cite

In this issue of JAMA Neurology, McKay and colleagues 1 found that adults with pediatric-onset multiple sclerosis (POMS) vs those with adult-onset MS (AOMS) performed more slowly on a widely used cognitive screening measure that tests information-processing speed. Patients were drawn from a Swedish nationwide cohort of 5704 adults with definite MS, of whom 300 had POMS.The Symbol Digit Modalities Test (SDMT) was serially administered during neurology visits. Early in their course, those with POMS performed somewhat better than those with AOMS. However, the 2 groups diverged by the time the POMS patients reached 30 years of age, with slower performance in the POMS group relative to the AOMS group, a difference that persisted over time. Although patients with POMS had greater disease duration, the findings remained even when disease duration was adjusted for in the model.Two other clinical research groups 2,3 have also reported a higher likelihood of cognitive impairment among adults with POMS vs AOMS, although on a smaller scale. Among patients with AOMS and POMS enrolled in a longitudinal study based in Boston, Massachusetts, the POMS group had similarly slower SDMT performances than the AOMS group, persisting when analyses controlled for age. 2 In an Italian study of adults that included a subgroup with POMS, 3 after adjusting for age at the time of testing, the POMS group was found to have more frequent cognitive impairment as defined with a broader neuropsychological test battery. 3 A methodological challenge in comparing cognitive performance in patients with POMS vs AOMS is that the developmental trajectory of cognitive processing speed is not well established. Younger individuals have faster cognitive processing speed performances than adults. Among those with MS, younger individuals may also have greater resilience and plasticity. However, these advantages may be outweighed by the accumulating effects of MS over time, as suggested by the findings in the study by McKay and colleagues 1 for those with POMS and a long disease duration. That is, despite initial resiliency and age-based advantages, the study's findings suggest greater deleterious long-term consequences from developing MS during a period of ongoing brain development.

show abstract

Patients with paediatric-onset multiple sclerosis are at higher risk of cognitive impairment in adulthood: An Italian collaborative study

Cited by 38 publications

References 33 publications

White matter plasticity and maturation in human cognition

White matter plasticity and maturation in human cognition

A systematic literature review of the burden of pediatric multiple sclerosis in North America and Europe

Long-term Cognitive Consequences for Patients With Pediatric-Onset Multiple Sclerosis

Contact Info

Product

Resources

About