Alpha-synuclein pathology in isolated rapid eye movement sleep behaviour disorder : a meta-analysis
© 2024 European Sleep Research Society..
Accumulating evidence indicates that patients with isolated rapid eye movement sleep behaviour disorder (iRBD), a prodromal stage of synucleinopathies, show abnormal deposition of misfolded alpha-synuclein (a-Syn) in peripheral tissues. The clinical utility of testing for a-Syn in iRBD is unclear. This meta-analysis focused on the utility of testing for the abnormal a-Syn phosphorylated at Ser129 (p-syn) and a-Syn seeding activity (a-Syn seed amplification assays [aSyn-SAA]). Following an electronic database search, 15 studies were included that provided at a minimum data on test positivity in participants with iRBD. Test positivity from cerebrospinal fluid (CSF) was 80% (95% confidence interval [CI] 68-88%, I2 = 71%) and for skin was 74.8% (95% CI 53.2-88.5%, I2 = 64%) for aSyn-SAA and 78.5% (95% CI 70.4-84.9%, I2 = 14%) for p-syn. The phenoconversion rate ratio of biopsy-positive versus biopsy-negative iRBD was 1.28 (95% CI 0.68-2.41, I2 = 0%). Skin as a source had a specificity of 99% (95% CI 95-100%, I2 = 0%; p = 0.01 compared to CSF). As a test, p-syn, had a specificity of 100% (95% CI 93-100%, I2 = 0%; p < 0.001) compared to aSyn-SAA. The odds ratio of a-Syn test positivity in iRBD versus other RBDs was 112 (95% CI 20-629, I2 = 0%). These results demonstrate clinically significant test positivity in iRBD and favour skin over CSF as the source of a-Syn pathological analysis, and p-syn over aSyn-SAA as the testing method. Overall, these findings indicate that testing for a-Syn could help in differentiating iRBD from RBD secondary to other conditions.
Medienart: |
E-Artikel |
---|
Erscheinungsjahr: |
2024 |
---|---|
Erschienen: |
2024 |
Enthalten in: |
Zur Gesamtaufnahme - year:2024 |
---|---|
Enthalten in: |
Journal of sleep research - (2024) vom: 08. Apr., Seite e14204 |
Sprache: |
Englisch |
---|
Beteiligte Personen: |
Iftikhar, Imran H [VerfasserIn] |
---|
Links: |
---|
Themen: |
Alpha‐synuclein seed amplification assay |
---|
Anmerkungen: |
Date Revised 08.04.2024 published: Print-Electronic Citation Status Publisher |
---|
doi: |
10.1111/jsr.14204 |
---|
funding: |
|
---|---|
Förderinstitution / Projekttitel: |
|
PPN (Katalog-ID): |
NLM370763076 |
---|
LEADER | 01000naa a22002652 4500 | ||
---|---|---|---|
001 | NLM370763076 | ||
003 | DE-627 | ||
005 | 20240408233658.0 | ||
007 | cr uuu---uuuuu | ||
008 | 240408s2024 xx |||||o 00| ||eng c | ||
024 | 7 | |a 10.1111/jsr.14204 |2 doi | |
028 | 5 | 2 | |a pubmed24n1369.xml |
035 | |a (DE-627)NLM370763076 | ||
035 | |a (NLM)38586895 | ||
040 | |a DE-627 |b ger |c DE-627 |e rakwb | ||
041 | |a eng | ||
100 | 1 | |a Iftikhar, Imran H |e verfasserin |4 aut | |
245 | 1 | 0 | |a Alpha-synuclein pathology in isolated rapid eye movement sleep behaviour disorder |b a meta-analysis |
264 | 1 | |c 2024 | |
336 | |a Text |b txt |2 rdacontent | ||
337 | |a ƒaComputermedien |b c |2 rdamedia | ||
338 | |a ƒa Online-Ressource |b cr |2 rdacarrier | ||
500 | |a Date Revised 08.04.2024 | ||
500 | |a published: Print-Electronic | ||
500 | |a Citation Status Publisher | ||
520 | |a © 2024 European Sleep Research Society. | ||
520 | |a Accumulating evidence indicates that patients with isolated rapid eye movement sleep behaviour disorder (iRBD), a prodromal stage of synucleinopathies, show abnormal deposition of misfolded alpha-synuclein (a-Syn) in peripheral tissues. The clinical utility of testing for a-Syn in iRBD is unclear. This meta-analysis focused on the utility of testing for the abnormal a-Syn phosphorylated at Ser129 (p-syn) and a-Syn seeding activity (a-Syn seed amplification assays [aSyn-SAA]). Following an electronic database search, 15 studies were included that provided at a minimum data on test positivity in participants with iRBD. Test positivity from cerebrospinal fluid (CSF) was 80% (95% confidence interval [CI] 68-88%, I2 = 71%) and for skin was 74.8% (95% CI 53.2-88.5%, I2 = 64%) for aSyn-SAA and 78.5% (95% CI 70.4-84.9%, I2 = 14%) for p-syn. The phenoconversion rate ratio of biopsy-positive versus biopsy-negative iRBD was 1.28 (95% CI 0.68-2.41, I2 = 0%). Skin as a source had a specificity of 99% (95% CI 95-100%, I2 = 0%; p = 0.01 compared to CSF). As a test, p-syn, had a specificity of 100% (95% CI 93-100%, I2 = 0%; p < 0.001) compared to aSyn-SAA. The odds ratio of a-Syn test positivity in iRBD versus other RBDs was 112 (95% CI 20-629, I2 = 0%). These results demonstrate clinically significant test positivity in iRBD and favour skin over CSF as the source of a-Syn pathological analysis, and p-syn over aSyn-SAA as the testing method. Overall, these findings indicate that testing for a-Syn could help in differentiating iRBD from RBD secondary to other conditions | ||
650 | 4 | |a Journal Article | |
650 | 4 | |a Review | |
650 | 4 | |a alpha‐synuclein seed amplification assay | |
650 | 4 | |a alpha‐synucleinopathy | |
650 | 4 | |a cerebrospinal fluid | |
650 | 4 | |a meta‐analysis | |
650 | 4 | |a phenoconversion | |
650 | 4 | |a rapid eye movment sleep behaviour disorder | |
700 | 1 | |a AlShimemeri, Sohaila |e verfasserin |4 aut | |
700 | 1 | |a Rabah, Hussein |e verfasserin |4 aut | |
700 | 1 | |a Rao, Saad Tauheed |e verfasserin |4 aut | |
700 | 1 | |a BaHammam, Ahmed S |e verfasserin |4 aut | |
773 | 0 | 8 | |i Enthalten in |t Journal of sleep research |d 1992 |g (2024) vom: 08. Apr., Seite e14204 |w (DE-627)NLM087669773 |x 1365-2869 |7 nnns |
773 | 1 | 8 | |g year:2024 |g day:08 |g month:04 |g pages:e14204 |
856 | 4 | 0 | |u http://dx.doi.org/10.1111/jsr.14204 |3 Volltext |
912 | |a GBV_USEFLAG_A | ||
912 | |a GBV_NLM | ||
951 | |a AR | ||
952 | |j 2024 |b 08 |c 04 |h e14204 |