Serum microRNA as indicators of Wolfram syndrome's progression in neuroimaging studies.
biomarkers
diabetes mellitus
genetic diseases
inborn
type 1
Journal
BMJ open diabetes research & care
ISSN: 2052-4897
Titre abrégé: BMJ Open Diabetes Res Care
Pays: England
ID NLM: 101641391
Informations de publication
Date de publication:
11 2020
11 2020
Historique:
received:
19
03
2020
revised:
14
09
2020
accepted:
14
09
2020
entrez:
2
11
2020
pubmed:
3
11
2020
medline:
27
5
2021
Statut:
ppublish
Résumé
Patients with the ultra-rare Wolfram syndrome (WFS) develop insulin-dependent diabetes and progressive neurodegeneration. The aim of the study was to quantify microRNAs (miRNAs) in sera from patients with WFS, correlate their expression with neurological imaging over time and compare miRNA levels with those observed in patients with type 1 diabetes mellitus (T1DM). We quantified miRNA expression (Qiagen, Germany) in two groups of patients: with WFS at study entry (n=14) and after 2 years of follow-up and in 15 glycated hemoglobin-matched (p=0.72) patients with T1DM. We observed dynamic changes in the expression of multiple miRNAs in patients with WFS parallel to disease progression and in comparison to the T1DM patients group. Among miRNAs that differed between baseline and follow-up WFS samples, the level of 5 increased over time (miR-375, miR-30d-5p, miR-30e-30, miR-145-5p and miR-193a-5p) and was inversely correlated with macular average thickness, while the expression of 2 (let-7g-5p and miR-22-3p) decreased and was directly correlated with neuroimaging indicators of neurodegeneration. Our findings show for the first time that serum miRNAs can be used as easily accessible indicators of disease progression in patients with WFS, potentially facilitating clinical trials on mitigating neurodegeneration.
Identifiants
pubmed: 33132210
pii: 8/2/e001379
doi: 10.1136/bmjdrc-2020-001379
pmc: PMC7607591
pii:
doi:
Substances chimiques
MIRN145 microRNA, human
0
MicroRNAs
0
Tungsten
V9306CXO6G
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Informations de copyright
© Author(s) (or their employer(s)) 2020. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.
Déclaration de conflit d'intérêts
Competing interests: None declared.
Références
Brain Res. 2019 Apr 1;1708:100-108
pubmed: 30552897
Sci Rep. 2019 Apr 12;9(1):6010
pubmed: 30979932
Oncotarget. 2018 Apr 3;9(25):17455-17465
pubmed: 29707120
Mol Neurobiol. 2019 Aug;56(8):5799-5814
pubmed: 30684217
Acta Diabetol. 2017 Nov;54(11):1019-1024
pubmed: 28856555
PLoS One. 2012;7(7):e40604
pubmed: 22792385
Mol Cell Biol. 2010 Sep;30(17):4197-210
pubmed: 20584986
J Alzheimers Dis Parkinsonism. 2018;8(1):
pubmed: 29862137
JAMA Neurol. 2017 Mar 1;74(3):275-285
pubmed: 28114622
Neurobiol Dis. 2015 Aug;80:42-53
pubmed: 25986729
Orphanet J Rare Dis. 2013 Apr 27;8:64
pubmed: 23981289
Int J Mol Sci. 2019 Nov 12;20(22):
pubmed: 31718095
J Mol Endocrinol. 2019 Aug;63(2):139-149
pubmed: 31277072
Biostatistics. 2007 Jan;8(1):118-27
pubmed: 16632515
J Proteome Res. 2017 Nov 3;16(11):4000-4008
pubmed: 28895401
Ann Neurol. 2013 Jun;73(6):729-40
pubmed: 23494648
PLoS One. 2013;8(1):e54222
pubmed: 23349832
Endokrynol Pol. 2014;65(4):295-7
pubmed: 25185852
Diabetologia. 2013 Jul;56(7):1512-9
pubmed: 23624530
Nature. 2004 Nov 11;432(7014):226-30
pubmed: 15538371
PLoS One. 2017 Dec 4;12(12):e0188980
pubmed: 29200427
Lancet. 1995 Dec 2;346(8988):1458-63
pubmed: 7490992
Diabetologia. 2012 Oct;55(10):2631-2635
pubmed: 22782286
Mol Cell. 2012 Nov 9;48(3):353-64
pubmed: 23022383
Diabetologia. 2016 Jul;59(7):1463-1473
pubmed: 27059371
Diabetes Care. 2011 Jul;34(7):1503-10
pubmed: 21602428
PLoS One. 2014 May 05;9(5):e94839
pubmed: 24797360
Hum Mol Genet. 2008 Dec 15;17(24):4012-21
pubmed: 18806274
Oncologist. 2010;15(7):673-82
pubmed: 20576643
Pediatr Diabetes. 2018 Dec;19(8):1407-1415
pubmed: 30259606
Diabetes Care. 2015 Mar;38(3):e36-7
pubmed: 25715420
BMC Pediatr. 2013 Aug 27;13:130
pubmed: 23981649
Biomarkers. 2016;21(2):129-37
pubmed: 26631297
Am J Med Genet A. 2007 Jul 15;143A(14):1605-12
pubmed: 17568405
Dev Med Child Neurol. 2019 Mar;61(3):359-365
pubmed: 30246501
Pediatr Diabetes. 2018 Feb;19(1):53-58
pubmed: 28436179
Proc Natl Acad Sci U S A. 2008 Jul 29;105(30):10513-8
pubmed: 18663219
Clin Endocrinol (Oxf). 2011 Nov;75(5):636-41
pubmed: 21564155
Neurol Neuroimmunol Neuroinflamm. 2019 Feb 14;6(2):e530
pubmed: 30800720
Aging Dis. 2019 Apr 1;10(2):329-352
pubmed: 31011481