Abstract

<h3>Objective:</h3> We investigated choroid plexus (CP) volume in pediatric multiple sclerosis (MS) patients compared to matched healthy controls (HC), possible sex-related effect, and the associations with clinical and structural MRI variables. <h3>Background:</h3> CP enlargement has been suggested as a reliable marker of neuroinflammation in adult MS patients, correlating with measures of brain structural damage and clinical disability. The presence and clinical relevance of CP enlargement in pediatric MS patients has not been evaluated yet. <h3>Design/Methods:</h3> Brain 3.0 T dual-echo and 3D T1-weighted sequences were identified retrospectively from 69 pediatric MS patients and 23 age- and sex-matched HC. CP volume was manually obtained from 3D T1-weighted scans by two expert raters. <h3>Results:</h3> CP segmentation was highly reproducible (intra-observer agreement: rater I=0.963, rater II=0.958; inter-observer agreement=0.968). Compared to HC, pediatric MS patients showed higher normalized CP volume (p&lt;0.001). When looking at sex-specific effect, both female and male pediatric MS patients showed higher normalized CP volume compared to sex-matched HC (females: p&lt;0.001; males: p=0.021), with a significant disease (MS vs HC) ‘ sex (females vs males) interaction (p=0.040). In MS patients, a higher normalized CP volume was significantly associated with higher brain T2-hyperintense white matter (WM) lesion volume (β=0.252, p=0.017), larger lateral ventricle volume (β=0.470, FDR p&lt;0.001), lower normalized brain volume (β=−0.413, FDR p=0.002) and lower normalized thalamic volume (β=0.291, FDR p=0.046). No associations with disease duration, EDSS score, normalized cortical and WM volumes were found (FDR p≥0.172). A significant effect of the disease in the negative association between normalized volumes of CP and thalami was found (FDR p=0.046). <h3>Conclusions:</h3> CP enlargement occurs in pediatric MS patients, suggesting its early involvement in the pathophysiology of the disease. The higher CP volume, that is found especially in female patients, supports the hypothesis of sex-related differences in the neuroinflammatory processes occurring already in pediatric MS patients. <b>Disclosure:</b> Monica Margoni has received research support from MAGNIMS. Dr. Gueye has nothing to disclose. Alessandro Meani has nothing to disclose. Elisabetta Pagani has nothing to disclose. Dr. Moiola has nothing to disclose. Mr. Preziosa has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Bristol Myers Squibb . Mr. Preziosa has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Sanofi Genzyme. Mr. Preziosa has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Novartis. Mr. Preziosa has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Roche. Mr. Preziosa has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Merck. Dr. Filippi has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Bayer, Biogen Idec, Merck-Serono, Novartis, Roche, Sanofi Genzyme, Takeda, and Teva Pharmaceutical Industries. Dr. Filippi has received personal compensation in the range of $5,000-$9,999 for serving on a Speakers Bureau for Bayer, Biogen Idec, Merck-Serono, Novartis, Roche, Sanofi Genzyme, Takeda, and Teva Pharmaceutical Industries. Dr. Filippi has received personal compensation in the range of $5,000-$9,999 for serving as an Editor, Associate Editor, or Editorial Advisory Board Member for Springer Nature. The institution of Dr. Filippi has received research support from Biogen Idec, Merck-Serono, Novartis, Roche, Teva Pharmaceutical Industries, Italian Ministry of Health, Fondazione Italiana Sclerosi Multipla, and ARiSLA (Fondazione Italiana di Ricerca per la SLA). Maria Assunta Rocca has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Bayer, Biogen, Bristol Myers Squibb, Celgene, Genzyme, Merck Serono, Novartis, Roche, and Teva. The institution of Maria Assunta Rocca has received research support from Italian Ministry of Health, MS Society of Canada and Fondazione Italiana Sclerosi Multipla.

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