Serial DaT-SPECT imaging in asymptomatic carriers of LRRK2 G2019S mutation: 8 years' follow-up
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URI: http://hdl.handle.net/10902/24042DOI: 10.1111/ene.15070
ISSN: 1351-5101
ISSN: 1468-1331
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Sánchez- Rodríguez, Antonio; Martínez-Rodríguez, Isabel; Sánchez-Juan, Pascual; Sierra, María; González-Aramburu, Isabel; Rivera-Sánchez, María; Andrés- Pacheco, Javier; Gutierrez-González, Ángela; García-Hernández, Adrián; Madera, Jorge; Delgado-Alvarado, Manuel; Infante Ceberio, JonFecha
2021-12Derechos
This is the peer reviewed version of the following article: [Sánchez-Rodríguez A, Martínez-Rodríguez I, Sánchez-Juan P, et al. Serial DaT-SPECT imaging in asymptomatic carriers of LRRK2 G2019S mutation: 8 years’ follow- up. Eur J Neurol. 2021;28:4204– 4208], which has been published in final form at [https://doi.org/10.1111/ene.15070 ]. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Use of Self-Archived Versions.
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European journal of neurology, 2021;28:4204? 4208
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Wiley
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Resumen/Abstract
Background:
Carriers of the G2019S mutation of LRRK2 provide a great opportunity to investigate the premotor stages of Parkinson's disease (PD). We have studied by serial clinical and dopamine transporter single photon emission computed tomography (DaT-SPECT) evaluations a cohort of asymptomatic carriers of the LRRK2-G2019S mutation in order to evaluate the usefulness of these tools as biomarkers. Here we report the results of the extended follow-up of this cohort at 8 years.
Methods:
Seventeen participants, of the 25 available from the 4-year evaluation, completed the 8-year assessment. UPDRS-III, UPSIT test and DaT-SPECT imaging (123I-ioflupane) were performed. We used repeated-measures linear mixed effects models to examine the changes in DaT binding over time.
Results:
Three carriers had converted to PD at 4 years. One additional carrier converted at 8 years. PD-converters had lower striatal DaT binding at baseline than non-converters. There was a significant decline of DaT binding over time, with a mean annual rate of 3.5%, with somewhat inter-individual and intra-individual variability and comparable between PD-converters and non-converters. No carrier with DAT binding ratio above an undefined threshold between 0.5 and 0.8 developed PD symptoms. The age-adjusted UPSIT score did not change significantly over time.
Conclusions:
The rate of conversion to PD at 8 years in this cohort aged ~58 years at baseline was 16%. The observed decline of DaT binding over time and its association with the phenotype render DaT-SPECT a potentially useful tool for monitoring the premotor stage of the disease, although at the individual level its ability to predict phenoconversion is limited.
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