BACKGROUND: Pediatric-onset multiple sclerosis (POMS) cases, defined as multiple sclerosis (MS) with onset before the age of 18, represent between 3 and 5 % of all MS patients. Anti-CD20 drugs mainly rituximab, ocrelizumab, and ofatumumab are being widely used in adult-onset MS. Their use in POMS is also being increasingly considered by experts.
OBJECTIVE: to review the latest evidence on safety and efficacy of the use of anti-CD20 therapies in POMS.
METHODS: An extensive search was performed in PubMed, Scopus, and Web of Science databases until the end of July 1st, 2024. Two independent reviewers screened the articles, and collected data. 832 studies were screened using Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) guidelines.
RESULTS: 12 studies on rituximab (328 patients) and 6 studies on ocrelizumab (106 patients) were synthesized. Using monoclonal antibodies in POMS patients has a noteworthy effect on reducing relapses and lesions and achieving no evidence of disease activity especially in highly active POMS patients. However, anti-CD20 therapies in MS are associated with potential adverse events (AEs). Additional data is required on the effect of anti-CD20 therapy on disability accrual.
CONCLUSIONS: Although anti-CD20 therapy is associated with some AEs, it can be provided in several circumstances, especially to patients with highly active disease, or ones resistant to platform therapies.

UNASSIGNED: Health disparities in adult-onset multiple sclerosis have been identified in the Black/African American (AA) population. A higher relapse rate has been suggested in Black/AA patients with pediatric-onset MS (POMS), but little work explores healthcare utilization and social determinants of health (SDOH).
UNASSIGNED: To evaluate racial, ethnic, and socioeconomic disparities in POMS outcomes.
UNASSIGNED: Retrospective chart review identified 31 eligible patients diagnosed with POMS at Children\'s of Alabama between 2013 and 2023. Demographics, outcomes, and healthcare utilization over 2 years from diagnosis were collected. Patient addresses were connected to SDOH measures from the US Census. Bivariate analysis was performed using Fisher\'s Exact Test, Wilcoxin Test, and 2-sided t-test.
UNASSIGNED: Black/AA children had a higher Expanded Disability Status Scale (EDSS) at first presentation (p = 0.0276) and were more likely to initiate fingolimod vs. glatiramer acetate (p = 0.0464). Living further from Children\'s of Alabama was associated with a higher most recent EDSS (p = 0.0301) and fewer neurology appointments (p = 0.0167). Families living in more socioeconomically deprived census tracts had significantly more hospital admissions.
UNASSIGNED: Black/AA POMS patients had a more severe initial presentation and were started on higher efficacy medication. We identified disparities in EDSS and healthcare utilization based on SDOH data linked to a child\'s home address.

BACKGROUND: Pediatric-onset Multiple Sclerosis (POMS) patients show more inflammatory disease compared with adult-onset MS. However, highly effective treatments are limited with only fingolimod being approved in Italy and natalizumab prescribed as off-label treatment.
OBJECTIVE: to compare the efficacy of natalizumab versus fingolimod in POMS.
METHODS: This is an observational longitudinal multicentre study including natalizumab- and fingolimod-treated POMS patients (N-POMS and F-POMS, respectively). We collected Annual Relapse Rate (ARR), Expanded Disability Status Scale (EDSS), Symbol Digit Modality Test (SDMT), and MRI activity at baseline (T0), 12-18 months (T1), and last available observation (T2).
RESULTS: We enrolled 57 N-POMS and 27 F-POMS patients from six Italian MS Centres. At T0, N-POMS patients showed higher ARR (p = 0.03), higher EDSS (p = 0.003) and lower SDMT (p = 0.04) at baseline compared with F-POMS. Between T0 and T1 ARR improved for both N-POMS and F-POMS (p < 0.001), while EDSS (p < 0.001) and SDMT (p = 0.03) improved only for N-POMS. At T2 (66.1 ± 55.4 months) we collected data from 42 out of 57 N-POMS patients showing no further ARR decrease.
CONCLUSIONS: Both natalizumab and fingolimod showed high and sustained efficacy in controlling relapses and natalizumab also associated to a disability decrease in POMS. This latter effect might be partly mediated by the high inflammatory activity at baseline in N-POMS.

Here we present a co-occurrence of a non-typical presentation of DIG/DIA and multiple sclerosis in a 13-year-old female. Our case highlights how a thorough investigation prior to treatment is needed in patients with such condition to choose proper management for better prognosis.

OBJECTIVE: We sought to explore the experiences and perceptions of the quality of life of adolescents with pediatric-onset multiple sclerosis and assess their readiness for academic, employment and/or health care-related transitions.
BACKGROUND: Adolescents with pediatric-onset multiple sclerosis face unique challenges in managing a chronic illness while navigating future scholastic, social and occupational goals. We conducted a qualitative study with in-depth, semi-structured interviews from July 2017 to March 2019. Adolescents with pediatric-onset multiple sclerosis were recruited from a pediatric neurology subspeciality practice until reaching data saturation. A total of 17 interviews were completed via telephone with participants ages 15 through 26.
RESULTS: Through content analysis of the interviews, we identified five major themes: (1) receiving a new diagnosis; (2) adapting to life with pediatric-onset multiple sclerosis; (3) evaluating education/career transition preparedness; (4) adjusting within family life and establishing support systems; and (5) assessing current medical services and preparedness for adult medical care.
CONCLUSIONS: Autonomy in health care management, adequate control of physical symptoms and sufficient family support impacted perceptions of quality of life. Implementing a dedicated transition visit, including the parent(s) of those with pediatric-onset multiple sclerosis, early in adolescence may provide an avenue for appropriate anticipatory guidance regarding available services, independent medical management and continuity of care.

Up to 10 years ago the most common approach to the treatment of pediatric MS (ped-MS) was to start with IFNB or GA (so-called first-line therapies or moderate-efficacy disease-modifying therapies [ME-DMTs]) and to switch to more aggressive treatments (or high-efficacy disease-modifying therapies [HE-DMTs]) in non-responder patients. The use of HE-DMTs as first choice was recommended in selected cases with an active, aggressive form of MS. Indications for the treatment of ped-MS were essentially derived from data of observational studies. Recently, results of three randomized clinical trials have been published as well as data from many observational studies evaluating the effect of new and more active DMTs, with clear evidence that HE-DMTs are more effective than ME-DMTs. Therefore, the paradigm of treatment for patients with MS onset before 18 years of age should be changed, offering treatment with HE-DMTs as first option, because of their superior effectiveness to prevent relapses and disease progression. HE-DMTs present an overall reassuring safety profile and obtain better adherence to treatment.

Some studies have suggested an inflammatory role of the choroid plexus (CP) in the pathophysiology of multiple sclerosis (MS), but mainly in adult patients. We aimed to evaluate clinical and MRI parameters in patients with pediatric-onset multiple sclerosis (POMS). We included 10 patients with POMS and 16 healthy controls (HC), evaluating clinical and neuroimaging variables (cerebral cortex, CP, deep gray matter structures, and demyelinating lesions). Most patients were girls (80%), with a mean age of 15.3 years. POMS individuals had a higher CP volume (p = 0.012) and lower thalamic volume (p = 0.038) compared to HC. This study shows an enlargement of the CP and lower thalamic volume in POMS patients compared to HC.

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BACKGROUND: Pediatric multiple sclerosis (PedMS) can hamper brain maturation. Aim of this study was to assess the neuropsychological profile of PedMS patients and their resting-state functional connectivity (RS FC).
METHODS: We assessed intelligence quotient (IQ), executive speed, and language in 76 PedMS patients. On a 3.0T scanner RS FC of brain networks was estimated with a seed-based analysis (subset of 58 right-handed PedMS patients and 22 matched healthy controls). Comparisons were run between controls and PedMS (whole cohort and by age).
RESULTS: Ninety-five% of patients had normal IQ. The highest rate of failure was observed in executive speed. PedMS showed reduced RS FC in all networks than controls, especially in the basal ganglia. In younger patients (<16-year-old, n = 32) reduced RS FC in the basal ganglia, language, and sensorimotor networks associated with poorer cognitive performance (p < 0.05; r range: 0.39; 0.56). Older patients (≥16-year-old, n = 26) showed increased RS FC in the basal ganglia, default-mode, sensorimotor, executive, and language networks, associated with poorer performance in executive speed and language abilities (p < 0.05; r range: -0.40; -0.59). In both groups, lower RS FC of the caudate nucleus associated with poorer executive speed.
CONCLUSIONS: The effect of PedMS on RS FC is clinically relevant and differs according to patients\' age.

BACKGROUND: Patient-powered research networks (PPRNs) for autoimmune disease are widely used in the adult population to recruit patients and drive patient-centered research, but few have included pediatric patients. We aimed to characterize viewpoints regarding research needs and participation in pediatric-onset multiple sclerosis (POMS) via a PPRN-disseminated survey.
METHODS: This is an exploratory, cross-sectional study. The study period was February 1, 2022, to February 9, 2023. Three questionnaires were disseminated to (1) patients with POMS (PwPOMS), (2) caregivers of PwPOMS (C-PwPOMS), and (3) health care providers/researchers in POMS (HR-POMS).
RESULTS: A total of 88 participants were included for analysis; 44% (n = 39) were PwPOMS, 42% (n = 37) were C-PwPOMS, and 14% (n = 12) were HR-POMS. Some PwPOMS (18%) and C-PwPOMS (9%) expressed research hesitancy, but more, 69% of PwPOMS and 68% of C-PwPOMS, were interested in research participation. Nevertheless, less than half of PwPOMS (38%) and C-PwPOMS (38%) reported previous research involvement. HR-POMS reported difficulties in funding (100%) and recruiting participants (58%). PwPOMS (67%), C-PwPOMS (62%), and HR-POMS (67%) were open to future involvement in PPRNs.
CONCLUSIONS: Participants with POMS in this study expressed strong interest in research involvement but also expressed participation hesitancy, which may contribute to recruiting challenges expressed by researchers. Although the exploratory design limits generalizability to the larger POMS population, this study shows PPRNs are well-suited to soliciting attitudes and opinions of key stakeholders in POMS. Future studies utilizing PPRNs for POMS should prioritize diverse, representative cohorts and focus on understanding and mitigating issues hindering research participation.