Memory, processing of emotional stimuli, and volume of limbic structures in pediatric-onset multiple sclerosis

Fabri, Tracy L.; Datta, Ritobrato; O’Mahony, Julia; Barlow-Krelina, Emily; Somma, Elisea De; Longoni, Giulia; Gur, Raquel E.; Gur, Ruben C.; Bacchus, Micky K.; Yeh, E. Ann; Banwell, Brenda; Till, Christine

doi:10.1016/j.nicl.2021.102753

Cited by 4 publications

(2 citation statements)

References 66 publications

(81 reference statements)

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“…8 This is an important observation in POMS but also monophasic ADS, where GM loss, specifically deep GM volume loss (e.g., thalamus), has been repeatedly shown as an important and early region of brain damage. 21,23-26 Importantly, the previous study on brain volume in pediatric ADS did not differentiate between MOG-positive and MOG-negative ADEM. 8 Although ADEM is primarily regarded as a WM disease, evidence from clinical, imaging, and neuropathologic studies suggests that cortical involvement is part of the disease process, which was particularly shown in association with MOG-abs.…”

Section: Discussionmentioning

confidence: 80%

Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis

Bartels

Baumgartner

Aigner

et al. 2023

Neurol Neuroimmunol Neuroinflamm

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Background and ObjectivesAcute disseminated encephalomyelitis (ADEM) is the most common phenotype in pediatric myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease. A previous study demonstrated impaired brain growth in ADEM. However, the effect of MOG antibodies on brain growth remains unknown. Here, we performed brain volume analyses in MOG-positive and MOG-negative ADEM at onset and over time.MethodsIn this observational cohort study, we included a total of 62 MRI scans from 24 patients with ADEM (54.2% female; median age 5 years), of which 16 (66.7%) were MOG positive. Patients were compared with healthy controls from the NIH pediatric MRI data repository and a matched local cohort. Mixed-effect models were applied to assess group differences and other relevant factors, including relapses.ResultsAt baseline and before any steroid treatment, patients with ADEM, irrespective of MOG antibody status, showed reduced brain volume compared with matched controls (median [interquartile range] 1,741.9 cm3 [1,645.1–1,805.2] vs 1,810.4 cm3[1,786.5–1,836.2]). Longitudinal analysis revealed reduced brain growth for both MOG-positive and MOG-negative patients with ADEM. However, MOG-negative patients showed a stronger reduction (−138.3 cm3[95% CI −193.6 to −82.9]) than MOG-positive patients (−50.0 cm3[−126.5 to −5.2]), independent of age, sex, and treatment. Relapsing patients (all MOG positive) showed additional brain volume loss (−15.8 cm3[−68.9 to 37.3]).DiscussionPatients with ADEM exhibit brain volume loss and failure of age-expected brain growth. Importantly, MOG-negative status was associated with a more pronounced brain volume loss compared with MOG-positive patients.

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Section: Discussionmentioning

confidence: 80%

Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis

Bartels

Baumgartner

Aigner

et al. 2023

Neurol Neuroimmunol Neuroinflamm

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“…This implies that the 22qDup group had more difficulty processing spatial information than information related to non‐verbal reasoning. Lastly, while there have been a handful of studies examining associations between brain structure and/or function and Penn‐CNB scores (Baller et al, 2021; Fabri et al, 2021; Huang et al, 2020; Khan et al, 2022; Prasad et al, 2022), and the tests have been validated with functional neuroimaging showing specificity of regional activation (Roalf et al, 2014), the precise neural correlates of each Penn‐CNB test has yet to be determined, which limits the ability to infer information about neurodevelopment from the results presented.…”

Section: Discussionmentioning

confidence: 99%

Distinct neurocognitive profiles and clinical phenotypes associated with copy number variation at the 22q11.2 locus

O'Hora,

Kushan‐Wells,

Schleifer

et al. 2023

Autism Research

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Rare genetic variants that confer large effects on neurodevelopment and behavioral phenotypes can reveal novel gene‐brain‐behavior relationships relevant to autism. Copy number variation at the 22q11.2 locus offer one compelling example, as both the 22q11.2 deletion (22qDel) and duplication (22qDup) confer increased likelihood of autism spectrum disorders (ASD) and cognitive deficits, but only 22qDel confers increased psychosis risk. Here, we used the Penn Computerized Neurocognitive Battery (Penn‐CNB) to characterized neurocognitive profiles of 126 individuals: 55 22qDel carriers (MAge = 19.2 years, 49.1% male), 30 22qDup carriers (MAge = 17.3 years, 53.3% male), and 41 typically developing (TD) subjects (MAge = 17.3 years, 39.0% male). We performed linear mixed models to assess group differences in overall neurocognitive profiles, domain scores, and individual test scores. We found all three groups exhibited distinct overall neurocognitive profiles. 22qDel and 22qDup carriers showed significant accuracy deficits across all domains relative to controls (episodic memory, executive function, complex cognition, social cognition, and sensorimotor speed), with 22qDel carriers exhibiting more severe accuracy deficits, particularly in episodic memory. However, 22qDup carriers generally showed greater slowing than 22qDel carriers. Notably, slower social cognition speed was uniquely associated with increased global psychopathology and poorer psychosocial functioning in 22qDup. Compared to TD, 22q11.2 copy number variants (CNV) carriers failed to show age‐associated improvements in multiple cognitive domains. Exploratory analyses revealed 22q11.2 CNV carriers with ASD exhibited differential neurocognitive profiles, based on 22q11.2 copy number. These results suggest that there are distinct neurocognitive profiles associated with either a loss or gain of genomic material at the 22q11.2 locus.

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The Impact of Neuroimmunologic Disease and Developing Nervous System

Santoro¹,

Jafarpour²,

Boyd³

et al. 2023

Pediatric Neurology

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Memory, processing of emotional stimuli, and volume of limbic structures in pediatric-onset multiple sclerosis

Cited by 4 publications

References 66 publications

Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis

Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis

Distinct neurocognitive profiles and clinical phenotypes associated with copy number variation at the 22q11.2 locus

The Impact of Neuroimmunologic Disease and Developing Nervous System

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