Cognitive and psychosocial features in childhood and juvenile MS

Amato, Maria Pia; Goretti, Benedetta; Ghezzi, Angelo; Lori, S.; Zipoli, Valentina; Moiola, Lucia; Falautano, Monica; De, Maria Fara; Viterbo, Rosa Gemma; Patti, Francesco; Vecchio, Rosario; Pozzilli, Carlo; Bianchi, Valentina; Roscio, Marco; Martinelli, Vittorio; Comi, Giancarlo; Portaccio, Emilio; Trojano, María

doi:10.1212/wnl.0b013e3181f4d821

Cited by 199 publications

(236 citation statements)

References 30 publications

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“…6,20,35,37,46 Investigations into the relationship between fatigue and neurocognitive functioning suggested that fatigue is unrelated to overall neurocognitive functioning. 6,[40][41][42] However, there was some evidence to suggest that fatigued caMS have impaired performance on some individual cognitive tasks, particularly motor-based tasks such as the Trail Making Test A and B, and on tasks requiring prolonged and complex mental activity, such as problem solving, complex attention and processing speed, and language comprehension tasks. 6,33 At present, the evidence does not substantially support a relationship between fatigue and any specific domain of cognitive functioning.…”

Section: Summary Of Evidencementioning

confidence: 99%

Understanding fatigue in paediatric multiple sclerosis: a systematic review of clinical and psychosocial factors

Carroll

Chalder

Hemingway

et al. 2015

Develop Med Child Neuro

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AIM Fatigue in children and adolescents with multiple sclerosis (caMS) is currently poorly understood. This review aimed to provide greater insight into this area and direction for future research by evaluating evidence of associations between fatigue and clinical, psychological, and social factors in caMS.METHOD Studies were identified by searching online databases, hand-searching reference lists, and requesting unpublished literature from key authors. Studies that examined fatigue in relation to at least one clinical, psychological, or social factor in caMS were included. Data on design, sample characteristics, measures of fatigue, clinical, psychological, and social variables, and key findings were extracted. Twelve studies were narratively synthesized.RESULTS Clinical factors appeared largely unrelated to fatigue, whereas associations between fatigue and tests of neurocognitive functioning, and fatigue and diagnosable psychiatric disorders, were mixed. However, fatigue and depressed mood consistently correlated. A small number of studies indicated associations between fatigue and reduced quality of life and school performance.INTERPRETATION A sufficient explanatory model of fatigue in caMS is lacking as studies in this area are few and diverse. Future research should endeavour to identify potentially modifiable clinical and psychosocial factors that are associated with fatigue in caMS so that interventions targeting such factors may be developed.Multiple sclerosis is a chronic neurodegenerative disorder characterized by inflammation and demyelination in the central nervous system, most often diagnosed in adulthood. 1 In rare cases, multiple sclerosis is diagnosed in childhood or adolescence.2 Paediatric acquired demyelinating syndromes, including paediatric multiple sclerosis, have an incidence rate of approximately 1 in 100 000. 3 In adult multiple sclerosis, sensory and motor impairments, optic neuritis, and bladder and bowel dysfunction are among the most common physical symptoms. In paediatrics, physical symptoms are less predominant but cognitive deficits more troublesome, typically affecting attention and processing speed, language, memory, and visuomotor functioning. [4][5][6] Moreover, psychosocial factors such as depression, lower quality of life, and fatigue are concerning features of paediatric multiple sclerosis, yet these features have received limited research attention to date. 6,7 Fatigue is the most common and one of the most disabling symptoms of multiple sclerosis, experienced by around 80% of adults with the disease. [8][9][10] Multiple sclerosis-related fatigue refers to a severe and overwhelming feeling of exhaustion and lack of energy, which may result in functional impairment.11 A growing body of evidence suggests that fatigue is also a significant problem in paediatric populations, affecting approximately 30% of the general adolescent population and a substantial proportion of children and adolescents with chronic illnesses such as cancer, rheumatoid arthritis, chronic pain, and neur...

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Section: Summary Of Evidencementioning

confidence: 99%

Understanding fatigue in paediatric multiple sclerosis: a systematic review of clinical and psychosocial factors

Carroll

Chalder

Hemingway

et al. 2015

Develop Med Child Neuro

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“…[3][4][5][6] Additionally, in POMS subjects, there is accumulating evidence of involvement of linguistic faculties 1,6 and lower intellectual efficiency in terms of intelligence quotient (IQ), particularly in those with younger age at MS onset. 7 An early MS disease onset can have a negative influence in school achievements and overall quality of life, [8][9][10][11] but it is unclear if it could lead to an increased risk for CI in later life. 3 Some studies suggest a negative impact of relapses, disease duration and physical disability in the cognitive performance of patients with POMS, 8,9,12 but these issues remain controversial.…”

Section: Introductionmentioning

confidence: 99%

“…3 Some studies suggest a negative impact of relapses, disease duration and physical disability in the cognitive performance of patients with POMS, 8,9,12 but these issues remain controversial. 10,11 There is little information about the long-term cognitive outcome in patients with POMS. Longitudinal studies published to date have relatively short followup periods (1-5 years), and suggest cognitive worsening, with variable rates of decline, although there are patients who may exhibit stable or improved cognitive function over time.…”

Section: Introductionmentioning

confidence: 99%

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

et al. 2017

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Background: Patients with paediatric-onset multiple sclerosis (POMS) could be at an increased risk for cognitive impairment (CI), given the potential harmful effects of disease activity in neurodevelopment. However, there is scarce information on their long-term cognitive outcomes. Objective: To compare the prevalence and profile of CI between adults with a history of POMS and those with classic, adult-onset multiple sclerosis (AOMS). Methods: Cognitive performance was assessed through the Brief Repeatable Battery (BRB) and the Stroop Test in consecutive patients referred to six Italian MS centres. CI was defined as impairment in ⩾2 cognitive domains. Results: In all, 119 patients with POMS and 712 with AOMS were included in this analysis. The prevalence of CI was 48.0% in AOMS, 44.5% in POMS; with similar neuropsychological profile between the two groups. However, when adjusting for current age, we found a significantly increased risk for CI (odds ratio (OR) = 1.71; p = 0.02) and for impairment in information processing speed (OR = 1.86; p < 0.01) in patients with POMS. A higher Expanded Disability Status Scale (EDSS) was also identified in POMS (p = 0.03) compared with AOMS patients. Conclusion: Patients with a history of POMS appear to be at higher risk of physical and cognitive disability than AOMS patients, after correcting for age effects, with particular involvement of information processing speed.

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“…The consequences of MS in a developing brain and early cognitive dysfunction are major concerns that can be evaluated only in children and cannot be extrapolated from studies in adults. 6,7,21,22 Cognitive function should be addressed in any therapeutic trial and requires the development of a core battery of neuropsychological tests applicable to international cross-sectional and longitudinal studies in pediatric MS. Challenges include differences in developmental trajectories and patterns of cognitive dysfunction in relation to patient age.…”

Section: Resultsmentioning

confidence: 99%

“…Current research is limited by variable tools used by different research groups, preventing pooling of data across studies, and limited availability of validated assessment tools with satisfactory norms. 6,[21][22][23] Requirements for cognitive batteries to be used in pediatric MS clinical trials are listed in table 3.…”

Section: Resultsmentioning

confidence: 99%

International Pediatric MS Study Group Clinical Trials Summit

et al. 2013

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Objective: Pediatric studies for new biological agents are mandated by recent legislation, necessitating careful thought to evaluation of emerging multiple sclerosis (MS) therapies in children with MS. Challenges include a small patient population, the lack of prior randomized clinical trials, and ethical concerns. The goal of this meeting was to assess areas of consensus regarding clinical trial design and outcome measures among academic experts involved in pediatric MS care and research.Methods: The Steering Committee of the International Pediatric MS Study Group identified key focus areas for discussion. A total of 69 meeting attendees were assembled, including 35 academic experts. Regulatory and pharmaceutical representatives also attended, and provided input, which informed academic expert consensus decisions. Results:The academic experts agreed that clinical trials were necessary in pediatric MS to obtain pharmacokinetic, safety and efficacy data, and regulatory approval allowing for greater medication access. The academic experts agreed that relapse was an appropriate primary outcome measure for phase III pediatric trials. An international standardized cognitive battery was identified. The pros and cons of various trial designs were discussed. Guidelines surrounding MRI studies, pharmacokinetics, pharmacodynamics, and registries were developed. The academic experts agreed that given the limited subject pool, a stepwise approach to the launch of clinical trials for the most promising medications is necessary in order to ensure study completion. Alternative approaches could result in unethical exposure of patients to trial conditions without gaining knowledge. Conclusion:Consensus points for conduct of clinical trials in the rare disease pediatric MS were identified amongst a panel of academic experts, informed by regulatory and industry stakeholders. There are limited studies informing the use of disease-modifying treatments in children with multiple sclerosis (MS). 1 No therapies are approved for pediatric MS by the US Food and Drug Administration (FDA), and only limited interferon use in adolescents with MS has been approved by the European Medicines Agency (EMA).1 As in adults, close to 40% of pediatric patients with MS discontinue treatment due to intolerance, toxicity, persisting relapses, or nonadherence, supporting the need for new therapeutic options in children.2 Several new molecules have shown efficacy in phase III trials and could be available in the near future for adults with MS.

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Cognitive and psychosocial features in childhood and juvenile MS

Abstract: Our findings confirm the importance of systematic assessment of cognitive and psychosocial issues in children and teens with MS. The progressive nature of the cognitive difficulties emphasizes the need for developing effective treatment strategies.

Cited by 199 publications

References 30 publications

Understanding fatigue in paediatric multiple sclerosis: a systematic review of clinical and psychosocial factors

Understanding fatigue in paediatric multiple sclerosis: a systematic review of clinical and psychosocial factors

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

International Pediatric MS Study Group Clinical Trials Summit

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