Serial DaT-SPECT imaging in asymptomatic carriers of LRRK2 G2019S mutation: 8 years' follow-up
| Autor: | María Sierra, Ángela Gutierrez-González, Jorge Madera, María Rivera-Sánchez, Jon Infante, Antonio Sánchez-Rodríguez, Adrián García-Hernández, Manuel Delgado-Alvarado, Isabel González-Aramburu, Pascual Sánchez-Juan, Isabel Martínez-Rodríguez, Javier Andrés-Pacheco |
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| Přispěvatelé: | Universidad de Cantabria |
| Rok vydání: | 2021 |
| Předmět: |
Oncology
medicine.medical_specialty Parkinson's disease Single-photon emission computed tomography Leucine-Rich Repeat Serine-Threonine Protein Kinase-2 G2019s mutation Internal medicine Medicine Humans Stage (cooking) Dopamine transporter Aged Tomography Emission-Computed Single-Photon Dopamine Plasma Membrane Transport Proteins medicine.diagnostic_test biology business.industry medicine.disease LRRK2 Neurology Cohort Mutation biology.protein Neurology (clinical) business Asymptomatic carrier Follow-Up Studies |
| Zdroj: | European journal of neurology, 2021;28:4204? 4208 UCrea Repositorio Abierto de la Universidad de Cantabria instname |
| ISSN: | 1468-1331 |
| Popis: | 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. |
| Databáze: | OpenAIRE |
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