Levodopa-induced dyskinesia in early-onset Parkinson's disease (EOPD) associates with glucocerebrosidase mutation: A next-generation sequencing study in EOPD patients in Thailand

PLoS One. 2023 Oct 31;18(10):e0293516. doi: 10.1371/journal.pone.0293516. eCollection 2023.

Abstract

Background: With the benefit of using next-generation sequencing (NGS), our aim was to examine the prevalence of known monogenic causes in early-onset Parkinson's disease (EOPD) patients in Thailand. The association between clinical features, such as levodopa-induced dyskinesia (LID), and genotypes were also explored.

Method: NGS studies were carried out for EOPD patients in the Tertiary-referral center for Parkinson's disease and movement disorders. EOPD patients who had LID symptoms were enrolled in this study (n = 47). We defined EOPD as a patient with onset of PD at or below 50 years of age. LID was defined as hyperkinetic movements including chorea, ballism, dystonia, myoclonus, or any combination of these movements resulting from levodopa therapy, which could be peak-dose, off-period, or diphasic dyskinesias.

Results: Pathogenic variants were identified in 17% (8/47) of the Thai EOPD patients, of which 10.6% (5/47) were heterozygous GBA variants (c.1448T>C in 3 patients and c.115+1G>A in 2 patients), 4.3% (2/47) homozygous PINK1 variants (c.1474C>T) and 2.1% (1/47) a PRKN mutation (homozygous deletion of exon 7). The LID onset was earlier in patients with GBA mutations compared to those without (34.8±23.4 vs 106.2±59.5 months after starting levodopa, respectively, p = 0.001). LID onset within the first 30 months of the disease was also found to be independently associated with the GBA mutation (odds ratio [95% confidence interval] = 25.00 [2.12-295.06], p = 0.011).

Conclusion: Our study highlights the high prevalence of GBA pathogenic variants in Thai patients with EOPD and the independent association of these variants with the earlier onset of LID. This emphasizes the importance of genetic testing in this population.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age of Onset
  • Dyskinesias* / etiology
  • Glucosylceramidase / genetics
  • Glucosylceramidase / therapeutic use
  • High-Throughput Nucleotide Sequencing
  • Homozygote
  • Humans
  • Levodopa / adverse effects
  • Levodopa / genetics
  • Mutation
  • Parkinson Disease* / drug therapy
  • Parkinson Disease* / epidemiology
  • Parkinson Disease* / genetics
  • Sequence Deletion
  • Thailand

Substances

  • Levodopa
  • Glucosylceramidase

Grants and funding

1. SBp, National Research Council of Thailand (NRCT); NRCT5-RSA63001-25, https://www.nrct.go.th/ 2. SBp, Thailand Science Research and Innovation Fund Chulalongkorn University (HEA663000054), https://www.tsri.or.th/ 3. SBp and ST, Ratchadapiseksompotch Fund (Faculty of Medicine, Chulalongkorn University); RA61/046, https://www.eng.chula.ac.th/wp-content/uploads/2021/07/res_cu_announcement_research-grant_2021_20210603.pdf 4. ST, Ratchadapiseksompotch Fund (Faculty of Medicine, Chulalongkorn University); GA64/11 and RA65/023, https://www.eng.chula.ac.th/wp-content/uploads/2021/07/res_cu_announcement_research-grant_2021_20210603.pdf The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.