Oana C Marian, Sophie Matis, Carol Dobson-Stone, Woojin S Kim, John B Kwok, Olivier Piguet, Glenda M Halliday, Ramon Landin-Romero, Anthony S Don
{"title":"额颞叶痴呆患者血浆己糖神经酰胺减少是白质完整性的生物标志物。","authors":"Oana C Marian, Sophie Matis, Carol Dobson-Stone, Woojin S Kim, John B Kwok, Olivier Piguet, Glenda M Halliday, Ramon Landin-Romero, Anthony S Don","doi":"10.1002/dad2.70131","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>Blood biomarkers are needed to facilitate new therapeutic trials and improve management of behavioral variant frontotemporal dementia (bvFTD). Since altered white matter integrity is characteristic of bvFTD, this study aimed to determine if plasma levels of myelin-enriched glycolipids are altered in bvFTD and correlate with white matter integrity.</p><p><strong>Methods: </strong>Nineteen glycolipids were quantified in bvFTD (<i>n</i> = 31) and control (<i>n</i> = 26) plasma samples. White matter integrity was assessed using magnetic resonance imaging (MRI)-derived fiber tract density and cross-section (FDC).</p><p><strong>Results: </strong>Eleven lipids were significantly lower in bvFTD compared to control subjects, and seven were inversely correlated with disease duration, with C22:0 hexosylceramide most strongly correlated. FDC was lower in frontotemporal white matter tracts of bvFTD compared to control subjects, and plasma C22:0 hexosylceramide was significantly correlated with FDC of these tracts in bvFTD but not control subjects.</p><p><strong>Discussion: </strong>Circulating glycolipids may be a valuable biomarker of myelin integrity and disease progression in FTD.</p><p><strong>Highlights: </strong>Blood biomarkers are needed for behavioral variant frontotemporal dementia (bvFTD).Plasma hexosylceramides are reduced in bvFTD cases compared with normal controls.Plasma hexosylceramides correlate with disease duration in bvFTD.Plasma hexosylceramides correlate with brain white matter integrity in bvFTD.Plasma glycolipids have potential as biomarkers of disease progression in bvFTD.</p>","PeriodicalId":53226,"journal":{"name":"Alzheimer''s and Dementia: Diagnosis, Assessment and Disease Monitoring","volume":"17 2","pages":"e70131"},"PeriodicalIF":4.0000,"publicationDate":"2025-06-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12136090/pdf/","citationCount":"0","resultStr":"{\"title\":\"Reduced plasma hexosylceramides in frontotemporal dementia are a biomarker of white matter integrity.\",\"authors\":\"Oana C Marian, Sophie Matis, Carol Dobson-Stone, Woojin S Kim, John B Kwok, Olivier Piguet, Glenda M Halliday, Ramon Landin-Romero, Anthony S Don\",\"doi\":\"10.1002/dad2.70131\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>Blood biomarkers are needed to facilitate new therapeutic trials and improve management of behavioral variant frontotemporal dementia (bvFTD). Since altered white matter integrity is characteristic of bvFTD, this study aimed to determine if plasma levels of myelin-enriched glycolipids are altered in bvFTD and correlate with white matter integrity.</p><p><strong>Methods: </strong>Nineteen glycolipids were quantified in bvFTD (<i>n</i> = 31) and control (<i>n</i> = 26) plasma samples. White matter integrity was assessed using magnetic resonance imaging (MRI)-derived fiber tract density and cross-section (FDC).</p><p><strong>Results: </strong>Eleven lipids were significantly lower in bvFTD compared to control subjects, and seven were inversely correlated with disease duration, with C22:0 hexosylceramide most strongly correlated. FDC was lower in frontotemporal white matter tracts of bvFTD compared to control subjects, and plasma C22:0 hexosylceramide was significantly correlated with FDC of these tracts in bvFTD but not control subjects.</p><p><strong>Discussion: </strong>Circulating glycolipids may be a valuable biomarker of myelin integrity and disease progression in FTD.</p><p><strong>Highlights: </strong>Blood biomarkers are needed for behavioral variant frontotemporal dementia (bvFTD).Plasma hexosylceramides are reduced in bvFTD cases compared with normal controls.Plasma hexosylceramides correlate with disease duration in bvFTD.Plasma hexosylceramides correlate with brain white matter integrity in bvFTD.Plasma glycolipids have potential as biomarkers of disease progression in bvFTD.</p>\",\"PeriodicalId\":53226,\"journal\":{\"name\":\"Alzheimer''s and Dementia: Diagnosis, Assessment and Disease Monitoring\",\"volume\":\"17 2\",\"pages\":\"e70131\"},\"PeriodicalIF\":4.0000,\"publicationDate\":\"2025-06-04\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12136090/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Alzheimer''s and Dementia: Diagnosis, Assessment and Disease Monitoring\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1002/dad2.70131\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/4/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q1\",\"JCRName\":\"CLINICAL NEUROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Alzheimer''s and Dementia: Diagnosis, Assessment and Disease Monitoring","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1002/dad2.70131","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/4/1 0:00:00","PubModel":"eCollection","JCR":"Q1","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
Reduced plasma hexosylceramides in frontotemporal dementia are a biomarker of white matter integrity.
Introduction: Blood biomarkers are needed to facilitate new therapeutic trials and improve management of behavioral variant frontotemporal dementia (bvFTD). Since altered white matter integrity is characteristic of bvFTD, this study aimed to determine if plasma levels of myelin-enriched glycolipids are altered in bvFTD and correlate with white matter integrity.
Methods: Nineteen glycolipids were quantified in bvFTD (n = 31) and control (n = 26) plasma samples. White matter integrity was assessed using magnetic resonance imaging (MRI)-derived fiber tract density and cross-section (FDC).
Results: Eleven lipids were significantly lower in bvFTD compared to control subjects, and seven were inversely correlated with disease duration, with C22:0 hexosylceramide most strongly correlated. FDC was lower in frontotemporal white matter tracts of bvFTD compared to control subjects, and plasma C22:0 hexosylceramide was significantly correlated with FDC of these tracts in bvFTD but not control subjects.
Discussion: Circulating glycolipids may be a valuable biomarker of myelin integrity and disease progression in FTD.
Highlights: Blood biomarkers are needed for behavioral variant frontotemporal dementia (bvFTD).Plasma hexosylceramides are reduced in bvFTD cases compared with normal controls.Plasma hexosylceramides correlate with disease duration in bvFTD.Plasma hexosylceramides correlate with brain white matter integrity in bvFTD.Plasma glycolipids have potential as biomarkers of disease progression in bvFTD.
期刊介绍：
Alzheimer''s & Dementia: Diagnosis, Assessment & Disease Monitoring (DADM) is an open access, peer-reviewed, journal from the Alzheimer''s Association® that will publish new research that reports the discovery, development and validation of instruments, technologies, algorithms, and innovative processes. Papers will cover a range of topics interested in the early and accurate detection of individuals with memory complaints and/or among asymptomatic individuals at elevated risk for various forms of memory disorders. The expectation for published papers will be to translate fundamental knowledge about the neurobiology of the disease into practical reports that describe both the conceptual and methodological aspects of the submitted scientific inquiry. Published topics will explore the development of biomarkers, surrogate markers, and conceptual/methodological challenges. Publication priority will be given to papers that 1) describe putative surrogate markers that accurately track disease progression, 2) biomarkers that fulfill international regulatory requirements, 3) reports from large, well-characterized population-based cohorts that comprise the heterogeneity and diversity of asymptomatic individuals and 4) algorithmic development that considers multi-marker arrays (e.g., integrated-omics, genetics, biofluids, imaging, etc.) and advanced computational analytics and technologies.
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