Catálogo de publicaciones - revistas

<jats:p>Objective</jats:p><jats:p>To explore and compare the use of health services in people with multiple sclerosis (MS) with and without fatigue.</jats:p><jats:p>Methods</jats:p><jats:p>Over a period of 30 months, the use of health services in 48 MS outpatients with persistent fatigue and 36 without fatigue was studied. Data were collected from a computerized register and by interviews, and analyzed with regard to disease severity categorized as mild or moderate/severe MS.</jats:p><jats:p>Results</jats:p><jats:p>Fatigued people with mild MS used more hospital outpatient care and primary care including rehabilitation, and a higher proportion had transportation service, compared with non-fatigued people with mild MS. In moderate/severe MS, the differences were that non-fatigued people used more occupational therapy in primary care and a higher proportion had salaried service. Regardless of MS severity, informal care was more common among fatigued people.</jats:p><jats:p>Conclusions</jats:p><jats:p>Overall, fatigued people with mild MS have more contacts with outpatient health care compared with non-fatigued people. There are few such differences in people with moderate/severe MS. The reasons for the differences in use between fatigued and non-fatigued people are not understood and need further exploration. Fatigued people more often receive informal care, thus support to caregivers are of particular importance if fatigue is present.</jats:p>

<jats:sec><jats:title>Background:</jats:title><jats:p> Several studies have shown that pregnancy reduces multiple sclerosis (MS) relapses, which increase in the early postpartum period. Postpartum relapse risk has been predicted by pre-pregnancy disease activity in some studies. </jats:p></jats:sec><jats:sec><jats:title>Objective:</jats:title><jats:p> To re-examine effect of pregnancy on relapses using the large international MSBase Registry, examining predictors of early postpartum relapse. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> An observational case–control study was performed including pregnancies post-MS onset. Annualised relapse rate (ARR) and median Expanded Disability Status Scale (EDSS) scores were compared for the 24 months pre-conception, pregnancy and 24 months postpartum periods. Clustered logistic regression was used to investigate predictors of early postpartum relapses. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> The study included 893 pregnancies in 674 females with MS. ARR (standard error) pre-pregnancy was 0.32 (0.02), which fell to 0.13 (0.03) in the third trimester and rose to 0.61 (0.06) in the first three months postpartum. Median EDSS remained unchanged. Pre-conception ARR and disease-modifying treatment (DMT) predicted early postpartum relapse in a multivariable model. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Results confirm a favourable effect on relapses as pregnancy proceeds, and an early postpartum peak. Pre-conception DMT exposure and low ARR were independently protective against postpartum relapse. This novel finding could provide clinicians with a strategy to minimise postpartum relapse risk in women with MS planning pregnancy. </jats:p></jats:sec>

<jats:sec><jats:title>Background:</jats:title><jats:p> Genetic ancestry, sex, and individual alleles have been associated with multiple sclerosis (MS) susceptibility. </jats:p></jats:sec><jats:sec><jats:title>Objective:</jats:title><jats:p> To determine whether established risk factors for disease onset are associated with relapse rate in pediatric MS. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> Whole-genome genotyping was performed for 181 MS or high-risk clinically isolated syndrome patients from two pediatric MS centers. Relapses and disease-modifying therapies were recorded as part of continued follow-up. Participants were characterized for 25-hydroxyvitamin D serum status. Ancestral estimates (STRUCTURE v2.3.1), human leukocyte antigen ( HLA) -DRB1*15 carrier status (direct sequencing), sex, and a genetic risk score (GRS) of 110 non-HLA susceptibility single-nucleotide polymorphisms (SNPs) were evaluated for association with relapse rate with Cox and negative binomial regression models. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> Over 622 patient-years, 408 relapses were captured. Girls had greater relapse rate than boys (incident rate ratio (IRR) = 1.40, 95% confidence interval (CI) = 1.04–1.87, p = 0.026). Participants were genetically diverse; ~40% ( N = 75) had &lt;50% European ancestry. HLA-DRB1*15 status modified the association of vitamin D status ( p<jats:sub>ixn</jats:sub> = 0.022) with relapse rate (per 10 ng/mL, in DRB1*15+ hazard ratio (HR) = 0.72, 95% CI = 0.58–0.88, p = 0.002; in DRB1*15− HR = 0.96, 95% CI = 0.83–1.12, p = 0.64). Neither European ancestry nor GRS was associated with relapse rate. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> We demonstrate that HLA-DRB1*15 modifies the association of vitamin D status with relapse rate. Our findings emphasize the need to pursue disease-modifying effects of MS genes in the context of environmental factors. </jats:p></jats:sec>

<jats:sec><jats:title>Background:</jats:title><jats:p> The study of cognitive reserve (CR) in relationship with cognitive impairment (CI) in pediatric-onset multiple sclerosis (POMS) may provide cues to identifying subjects at higher risk of impairment and scope for therapeutic strategies. </jats:p></jats:sec><jats:sec><jats:title>Objectives:</jats:title><jats:p> To assess the potential impact of CR on cognition in a cohort of POMS patients. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> In all, 48 POMS patients were followed up for 4.7 ± 0.4 years. CI was defined as the failure of ⩾3 tests on an extensive neuropsychological battery. Change of neuropsychological performance was assessed through the Reliable Change Index (RCI) method. At baseline, CR was estimated by measuring the intelligence quotient (IQ). The relationships were assessed through multivariable regression analyses. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> At baseline, CI was detected in 14/48 (29.2%) patients. Two out of 57 healthy control (HC; 3.5%) met the same criteria of CI ( p &lt; 0.001). A deteriorating cognitive performance using the RCI method was observed in 18/48 patients (37.6%). Among the 34 cases who were cognitively preserved at baseline, a higher reserve predicted stable/improving performance (odds ratio (OR) = 1.11; 95% confidence interval (CI): 1.03–1.20; p = 0.006). </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Our results suggest that higher CR in POMS patients may protect from CI, particularly in subjects with initial cognitive preservation, providing relevant implications for counseling and rehabilitation strategies. </jats:p></jats:sec>

<jats:sec><jats:title>Objective:</jats:title><jats:p> To assess retinal ganglion cell (RGC) injury and sex differences in axon loss in pediatric multiple sclerosis (MS). </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> This is a cross-sectional evaluation of consecutive pediatric MS subjects and controls. Eyes with acute optic neuritis (ON) within 6 months of visit were excluded. Spectral domain optical coherence tomography (OCT) included peripapillary ring and macular scans with post-acquisition segmentation of retinal layers using automated software (Heidelberg v1.8.6.0). Generalized estimating equations (GEEs) measured associations of sex, history of ON, disease duration, and age with OCT outcomes. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> In all, 53 MS subjects (100 eyes, median disease duration = 1.0 years, interquartile range (IQR) = 0.3, 2.5) were compared to 19 control subjects (38 eyes). Eyes with history of ON showed reduced retinal nerve fiber layer (RNFL: −26.8 µm, 95% confidence interval (CI) = −38.9, −14.8, p &lt; 0.001) and 26% lower ganglion cell layer (GCL) volumes (−0.12 mm<jats:sup>3</jats:sup>, 95% CI = −0.16, −0.072, p &lt; 0.001) compared to control eyes. Non-ON MS eyes had lower temporal RNFL (−11.9 µm, 95% CI = −18.6, −5.3, p &lt; 0.001) and GCL volumes (−0.036 mm<jats:sup>3</jats:sup>, 95% CI = −0.06, −0.011, p = 0.004) than control eyes. In MS eyes, males versus females had lower global RNFL (−9.4 µm, 95% CI = −17.4, −1.33, p = 0.022) and in ON eyes had lower temporal quadrant RNFL (−9.6 µm, 95% CI = −15.1, −4.15, p = 0.001). </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Subclinical retinal injury occurs in pediatric-onset MS patients without a history of ON. As in adult-onset MS, substantial GCL thinning is present in eyes with prior ON. Finally, greater retinal axonal injury occurs in boys compared to girls. </jats:p></jats:sec>

<jats:sec><jats:title>Background:</jats:title><jats:p> The risk factors for conversion from relapsing-remitting to secondary progressive multiple sclerosis remain highly contested. </jats:p></jats:sec><jats:sec><jats:title>Objective:</jats:title><jats:p> The aim of this study was to determine the demographic, clinical and paraclinical features that influence the risk of conversion to secondary progressive multiple sclerosis. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> Patients with adult-onset relapsing–remitting multiple sclerosis and at least four recorded disability scores were selected from MSBase, a global observational cohort. The risk of conversion to objectively defined secondary progressive multiple sclerosis was evaluated at multiple time points per patient using multivariable marginal Cox regression models. Sensitivity analyses were performed. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> A total of 15,717 patients were included in the primary analysis. Older age (hazard ratio (HR) = 1.02, p &lt; 0.001), longer disease duration (HR = 1.01, p = 0.038), a higher Expanded Disability Status Scale score (HR = 1.30, p &lt; 0.001), more rapid disability trajectory (HR = 2.82, p &lt; 0.001) and greater number of relapses in the previous year (HR = 1.07, p = 0.010) were independently associated with an increased risk of secondary progressive multiple sclerosis. Improving disability (HR = 0.62, p = 0.039) and disease-modifying therapy exposure (HR = 0.71, p = 0.007) were associated with a lower risk. Recent cerebral magnetic resonance imaging activity, evidence of spinal cord lesions and oligoclonal bands in the cerebrospinal fluid were not associated with the risk of conversion. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Risk of secondary progressive multiple sclerosis increases with age, duration of illness and worsening disability and decreases with improving disability. Therapy may delay the onset of secondary progression. </jats:p></jats:sec>

<jats:sec><jats:title>Background:</jats:title><jats:p> Individuals with multiple sclerosis (MS) regularly report making strategic decisions about whether to share their diagnosis or keep it a secret; for many, this represents a key stressor. Surprisingly, the impact of disclosure and concealment are understudied in MS and a formal measurement instrument is lacking. We developed the Diagnosis Disclosure and Concealment in MS (DISCO-MS) survey, a self-assessment tool evaluating (1) frequency of concealment behaviors and (2) expected consequences of diagnosis disclosure in persons with MS. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> A systematic mixed-methods process was used for the design and initial validation of the DISCO-MS. Associations of DISCO-MS responses to demographics, clinical variables, and mood were examined in 204 participants with MS. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> The 39-item DISCO-MS shows good psychometric characteristics. Approximately 25% of respondents conceal their diagnosis, particularly in professional settings. Higher concealment behaviors were associated with younger age, shorter disease duration, and lower physical disability. Nearly 50% of respondents believed that talking openly about their diagnosis might have undesirable professional and interpersonal consequences. Younger age, higher depression, and higher anxiety were associated with greater expectations of negative consequences. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Development and validation of the DISCO-MS presents the first step toward systematic study of the impact of DISCO on people with MS. </jats:p></jats:sec>

<jats:sec><jats:title>Background:</jats:title><jats:p> Multiple sclerosis (MS) is commonly associated with decision-making, neurocognitive impairments, and mood and motivational symptoms. However, their relationship may be obscured by traditional scoring methods. </jats:p></jats:sec><jats:sec><jats:title>Objectives:</jats:title><jats:p> To study the computational basis underlying decision-making impairments in MS and their interaction with neurocognitive and neuropsychiatric measures. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> Twenty-nine MS patients and 26 matched control subjects completed a computer version of the Iowa Gambling Task (IGT). Participants underwent neurocognitive evaluation using an expanded version of the Brief Repeatable Battery. Hierarchical Bayesian Analysis was used to estimate three established computational models to compare parameters between groups. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> Patients showed increased learning rate and reduced loss-aversion during decision-making relative to control subjects. These alterations were associated with: (1) reduced net gains in the IGT; (2) processing speed, executive functioning and memory impairments; and (3) higher levels of depression and current apathy. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Decision-making deficits in MS patients could be described by the interplay between latent computational processes, neurocognitive impairments, and mood/motivational symptoms. </jats:p></jats:sec>

<jats:p> Over the recent years, the treatment of multiple sclerosis (MS) and neuromyelitis optica spectrum disorder (NMOSD) has evolved very rapidly and a large number of disease-modifying treatments (DMTs) are now available. However, most DMTs are associated with adverse events, the most frequent of which being infections. Consideration of all DMT-associated risks facilitates development of risk mitigation strategies. An international focused workshop with expert-led discussions was sponsored by the European Committee for Treatment and Research in Multiple Sclerosis (ECTRIMS) and was held in April 2021 to review our current knowledge about the risk of infections associated with the use of DMTs for people with MS and NMOSD and corresponding risk mitigation strategies. The workshop addressed DMT-associated infections in specific populations, such as children and pregnant women with MS, or people with MS who have other comorbidities or live in regions with an exceptionally high infection burden. Finally, we reviewed the topic of DMT-associated infectious risks in the context of the current SARS-CoV-2 pandemic. Herein, we summarize available evidence and identify gaps in knowledge which justify further research. </jats:p>

<jats:sec><jats:title>Background:</jats:title><jats:p> Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) may be associated with relapsing disease, but clinical progression independent of relapse activity is rare. </jats:p></jats:sec><jats:sec><jats:title>Objectives:</jats:title><jats:p> To report progressive disease in a patient with MOGAD. </jats:p></jats:sec><jats:sec><jats:title>Methods:</jats:title><jats:p> A single retrospective case report. </jats:p></jats:sec><jats:sec><jats:title>Results:</jats:title><jats:p> At 4 years of age, the patient had a single episode of acute disseminated encephalomyelitis. She remained well until age 17 years but over the next 9 years developed progressive spastic quadriparesis, cognitive and bulbar dysfunction. Brain imaging showed a leukodystrophy-like pattern of white matter abnormality with contrast enhancement at different time points. Myelin oligodendrocyte glycoprotein (MOG)-IgG was repeatedly positive by live cell-based assay. </jats:p></jats:sec><jats:sec><jats:title>Conclusion:</jats:title><jats:p> Secondary progression may be a rare presentation of MOG-IgG-associated disease. </jats:p></jats:sec>