Leukocyte Telomere Length as Potential Biomarker of HD Progression: A Follow-Up Study.
Huntington’s disease
fluid biomarkers
leukocyte telomere length
neurodegenerative diseases
Journal
International journal of molecular sciences
ISSN: 1422-0067
Titre abrégé: Int J Mol Sci
Pays: Switzerland
ID NLM: 101092791
Informations de publication
Date de publication:
03 Nov 2022
03 Nov 2022
Historique:
received:
17
10
2022
revised:
28
10
2022
accepted:
29
10
2022
entrez:
11
11
2022
pubmed:
12
11
2022
medline:
15
11
2022
Statut:
epublish
Résumé
The identification of biomarkers for neurodegenerative disorders such as Huntington's disease (HD) is crucial for monitoring disease progression and therapeutic trial outcomes, especially in the pre-manifest disease stage (pre-HD). In a previous study, we observed that leukocyte telomere length (LTL) was strongly correlated with the estimated time to clinical onset in pre-HD subjects. To validate this hypothesis, we designed a follow-up study in which we analyzed LTL in 45 pre-HD stage subjects at baseline (T0) and then again after clinical onset at follow-up (T1); the follow-up interval was about 3 years, and the CAG range was 39-51 repeats; 90 peripheral blood mononuclear cell samples (PBMCs) were obtained from the Enroll-HD biorepository. In pre-HD subjects at T0, LTL was significantly reduced by 22% compared to the controls and by 14% from T0 at T1. No relationship was observed between the LTL and CAG numbers in subjects carrying different CAG repeats at T0 and at T1, suggesting that LTL reduction occurs independently of CAG number in pre-HD subjects. ROC curve analysis was used to test the validity of LTL as a potential biomarker of HD progression and showed that LTL measurement is extremely accurate in discriminating pre-HD subjects from the controls and even pre-HD from manifest HD, thus yielding a robust prognostic value in pre-HD subjects.
Identifiants
pubmed: 36362235
pii: ijms232113449
doi: 10.3390/ijms232113449
pmc: PMC9654348
pii:
doi:
Substances chimiques
Biomarkers
0
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Subventions
Organisme : Sapienza University of Rome
ID : 2018; 2019
Organisme : EHDN seed fund
ID : grant 0942
Organisme : AICH
ID : 2020
Organisme : Fondo Ordinario Enti (FOE D.M 865/2019) in the framework of a collaboration agreement between CNR and EBRI
ID : 2019-2021
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