Randomized controlled trial of intravenous immunoglobulin for autoimmune postural orthostatic tachycardia syndrome (iSTAND).

OBJECTIVE: This study assesses response to intravenous immunoglobulin (IVIG) in presumed autoimmune postural orthostatic tachycardia syndrome (POTS). BACKGROUND: POTS may be associated with autoimmune disorders, serum autoantibodies, or recent infection. Uncontrolled case studies suggest that IVIG is beneficial for treating autoimmune POTS. No previous randomized controlled trials have been conducted. METHODS: This single-site randomized controlled trial compared IVIG with intravenous albumin infusions. Albumin comparator ensured blinding and control for effects of volume expansion. Eligible patients with POTS had COMPASS-31 total weighted score ≥ 40 and met predetermined criteria suggesting autoimmunity. Over 12 weeks, participants received eight infusions (0.4 gm/kg each). Four infusions were given weekly followed by four infusions every other week. Primary outcome measure was improvement in COMPASS-31 2 weeks after final infusion. RESULTS: A total of 50 participants consented; 30 met inclusion criteria and received study drug (16 IVIG and 14 albumin; 29 female). Group baseline characteristics were well matched; 27 participants completed treatment protocol. Change in COMPASS-31 did not differ between groups (median change [IQR]; IVIG: -5.5 [-23.3, 2.5] versus albumin: -10.6 [-14.1, -4.7]; p-value = 0.629). The IVIG group had a higher response rate (46.7% versus 38.5%), but this was not statistically significant. Adverse events were common but usually mild and did not differ between treatment groups. CONCLUSIONS: This small randomized controlled trial of IVIG in POTS found no statistical difference in response compared with albumin infusion. Both groups showed improvement possibly related to volume expansion or other effects obscuring group differences. These findings inform development of future immunomodulatory clinical trials in POTS.

Clinical analysis of myelin oligodendrocyte glycoprotein antibody-associated disease in a diverse cohort of children: A single-center observational study.

BACKGROUND: Prognostic markers for relapse and neurological disability following the first clinical event in children with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) remain lacking. We investigated the clinical profiles and early prognostic factors associated with relapsing disease or impaired functional outcome in a large single-center cohort of pediatric MOGAD. METHODS: We retrospectively analyzed the clinical and paraclinical data and treatment outcomes of children with MOGAD seen at Children's Health in Dallas, Texas from 2009 to 2022. Univariate analyses were used to evaluate factors from initial event associated with relapsing disease course and impaired functional outcome (modified Rankin scale [mRS] >1) at final follow-up. RESULTS: Our cohort comprised of 87 children of diverse race/ethnicity. Presentation with acute disseminated encephalomyelitis (ADEM) was more frequent in children aged ≤8 years and Caucasian background, whereas presentation with optic neuritis was more common in children aged >8 years and other race/ethnicity. 44.3 % (27/61) had relapsing disease course, of whom 48.0 % had multiple relapses. 30.3 % (23/76) had final mRS >1. Children with abnormal electroencephalogram had reduced relapse risk. Children with ADEM presentation, severe disease, low MOG-IgG titer, and central and systemic inflammation (represented by cerebrospinal fluid pleocytosis and serum leukocytosis, respectively) at onset had higher likelihood of final mRS >1. CONCLUSION: Abnormal electroencephalogram at the first event was associated with reduced relapse risk while disease severity and peripheral inflammation significantly contributed to final neurological disability. Further studies are needed to validate these findings as early risk factors for disability and relapse and to identify optimal treatment strategies.

Increased healthcare utilization in the year before multiple sclerosis diagnosis.

BACKGROUND: Studies have suggested possible multiple sclerosis (MS) prodrome with non-routine healthcare utilization as an indicator. The objective of this project was to compare utilization in the four years (years -1, -2, -3, -4) before clinically definite diagnosis and examine demographic associations. METHODS: i2B2 database search at the Medical College of Wisconsin Comprehensive MS center yielded 613 patients between 07/01/2018 and 07/01/2022. Patients with established MS diagnosis, MS mimicker diagnoses, and pediatric-onset MS were excluded; 108 met the criteria for adult patients ≥ 18 years of age newly diagnosed with clinically definite MS after manual chart review. Utilization score for each of the four years before diagnosis was calculated; demographic variables were also collected. Adjusted repeated measures mixed model and Pearson correlation analysis were performed; P value was set at <0.05 for statistical significance. RESULTS: Utilization was greatest for year -1 compared to years -2, -3, and -4 after demographics adjustment (p < 0.001). Utilization was greater (p < 0.05) for older age and unmarried, patients. CONCLUSIONS: Utilization increased a year before formal MS diagnosis, suggestive of prodromal presentation.

Medical Student Perceptions of Sociocultural Issues in Healthcare: A Multisite Study of Medical Spanish Education.

THEORY: Cultural competence and humility are core elements of medical education in a diverse society. Language is inseparable from culture, as language informs, indexes, frames, and encodes both culture and worldview. Spanish is the most common non-English language taught in U.S. medical schools, yet medical Spanish courses tend to artificially separate language from culture. It is unknown to what extent medical Spanish courses advance students' sociocultural knowledge or patient care skills. HYPOTHESES: Based on current predominant pedagogy, medical Spanish classes may not adequately integrate sociocultural issues relevant to Hispanic/Latinx health. We hypothesized that students who completed a medical Spanish course would not demonstrate significant gains in sociocultural skills following the educational intervention. METHOD: An interprofessional team developed a sociocultural questionnaire, and 15 medical schools invited their students to complete the questionnaire before and after completing a medical Spanish course. Of participating schools, 12 implemented a standardized medical Spanish course and three served as control sites. Survey data were analyzed regarding: (1) perceived sociocultural competence (recognition of common cultural beliefs, recognition of culturally normative non-verbal cues, gestures, and social behaviors, ability to address sociocultural issues in healthcare context, and knowledge of health disparities); (2) application of sociocultural knowledge; and (3) demographic factors and self-rated language proficiency (Poor, Fair, Good, Very Good, or Excellent) on the Interagency Language Roundtable scale for healthcare (ILR-H). RESULTS: Overall, 610 students participated in sociocultural questionnaire between January 2020 and January 2022. After the course, participants reported an increased understanding of cultural aspects of communication with Spanish-speaking patients and the ability to apply sociocultural knowledge to patient care (all p < 0.001). When analyzed by demographic factors, students who identified as Hispanic/Latinx or as heritage speakers of Spanish tended to report increased sociocultural knowledge/skills following the course. When examined by Spanish proficiency, preliminary trends showed that students at both ILR-H Poor and Excellent levels did not demonstrate gains in sociocultural knowledge or ability to apply sociocultural skills. Students at sites with a standardized course were likely to improve sociocultural skills in mental health conversations (p < 0.001) while students at control sites were not (p = 0.05). CONCLUSIONS: Medical Spanish educators may benefit from additional guidance on teaching sociocultural aspects of communication. Our findings support that students at ILR-H levels of Fair, Good, and Very Good are particularly well-suited for gaining sociocultural skills in current medical Spanish courses. Future studies should explore potential metrics to evaluate cultural humility/competence within actual interactions with patients.