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International Parkinson and Movement Disorder Society
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        VOLUME 28, ISSUE 4 • DECEMBER 2024.  Full issue »

2024’s most impactful clinical neuroscience studies

During the 2024 MDS International Congress, Dr Hugo Morales-Briceno discussed the most clinically impactful articles of the past 12 months in the Highlights session.  


 

Parkinson’s disease  

Parkinson’s disease (PD) is a progressive neurodegenerative disorder with no current treatments to slow its progression. The 2019 LEAP study explored the effects of delaying levodopa treatment but found no significant differences in disease progression. In the follow-up five-year analysis (1), the prevalence of motor fluctuations including dyskinesia was not different between groups, suggesting that the timing of levodopa initiation does not reduce the risk of motor fluctuations, highlighting disease duration as a stronger factor.  

Dementia is a significant concern in PD, with early studies estimating a 46-58% likelihood of dementia after 10 years. However, more recent research, including the PPMI and Pennsylvania University cohorts, found much lower incidences at 10 years—8.5% and 27%, respectively (2). Nevertheless, such variation in dementia rates between these studies is unknown.  

The PD GENEration and ROPAD studies, which assessed large PD cohorts, found that 13-15% of patients had identifiable genetic mutations, with GBA and LRRK2 being the most common (3, 4). Patients with early onset, affected family members, or high-risk ancestry had a higher rate of genetic diagnoses. Recessive mutations in the PRKN gene are linked to young-onset PD, however some mutations can be missed by conventional testing. Long-read sequencing has improved the diagnosis of patients with only one PRKN mutation, potentially increasing the diagnostic yield in future studies (5, 6). 

In terms of treatment, levodopa infusion therapies show promise for managing motor fluctuations in PD (7, 8). The BouNDless trial demonstrated that continuous subcutaneous levodopa-carbidopa infusion (ND0612) improved ON time without troublesome dyskinesias by 1.72-hours and reduced OFF time by 1.4-hours. Another trial of subcutaneous foslevodopa/foscarbidopa showed similar improvements in ON and OFF times, though infusion-site reactions remain a challenge. 

Atypical Parkinsonism 

A study of pathological proven MSA cases found that while MRI criteria did not improve diagnostic sensitivity, clinical criteria alone had better diagnostic accuracy (9). However, the low sensitivity of early MSA diagnosis suggests the need for additional biomarkers. 

Deep Brain Stimulation for dystonia  

Deep brain stimulation (DBS) has shown promise in treating paediatric dystonia. A systematic review indicated a ~60% improvement in dystonia scores after one year of GPi-DBS, with the best outcomes in patients with TOR1A, SCGE, KMT2B, and idiopathic dystonia (10). Bilateral GPi-DBS has also proven effective in treating status dystonicus, with a lower mortality rate compared to pharmacotherapy (11). 

Genetics in Movement Disorders  

Paroxysmal Kinesigenic Dyskinesias are commonly caused by PRRT2 mutations. Two recent studies have independently identified dominant KCNJ10 variants as a novel cause of PKD (12, 13). Recessive KCNJ10 cause EAST syndrome (epilepsy, ataxia, sensorineural deafness, seizures and tubulopathy), and interestingly, PKD patients due to KCNJ10 may have some attenuated features of EAST syndrome. Intronic FGF14 expansions is one of the most common causes of late onset ataxia. Recent studies have highlighted the slow progression nature, but also presentations as episodic ataxia in up to 13% (14). A recent study reported that 75% of FGF14 patients treated with 4-aminopyridine improve the frequency and intensity of episodic symptoms (15). 

 

References 

  1. Frequin HL, Verschuur CVM, Suwijn SR, et al. Long-Term Follow-Up of the LEAP Study: Early Versus Delayed Levodopa in Early Parkinson's Disease. Movement Disorders. 2024;39(6):975-982.

  2. Gallagher J, Gochanour C, Caspell-Garcia C, et al. Long-Term Dementia Risk in Parkinson Disease. Neurology. 2024;103(5):e209699. 

  3. Cook L, Verbrugge J, Schwantes-An TH, et al. Parkinson's disease variant detection and disclosure: PD GENEration, a North American study. Brain. 2024;147(8):2668-2679. 

  4. Westenberger A, Skrahina V, Usnich T, et al. Relevance of genetic testing in the gene-targeted trial era: the Rostock Parkinson's disease study. Brain. 2024 Aug 1;147(8):2652-2667.  

  5. Daida K, Funayama M, Billingsley KJ, et al. Long-Read Sequencing Resolves a Complex Structural Variant in Parkinson's Disease. Movement Disorders. 2023;38(12):2249-2257. 

  6. Cogan G, Daida K, Billingsley KJ, et al. Long-Read Sequencing Unravels the Complexity of Structural Variants in in Two Individuals with Early-Onset Parkinson's Disease. Movement Disorders. 2024;39(9):1647-1648. 

  7. Espay AJ, Stocchi F, Pahwa R, et al. Safety and efficacy of continuous subcutaneous levodopa–carbidopa infusion (ND0612) for Parkinson's disease with motor fluctuations (BouNDless): a phase 3, randomised, double-blind, double-dummy, multicentre trial. The Lancet Neurology. 2024;23(5):465-476. 

  8. Aldred J, Freire-Alvarez E, Amelin AV, et al. Continuous Subcutaneous Foslevodopa/Foscarbidopa in Parkinson’s Disease: Safety and Efficacy Results From a 12-Month, Single-Arm, Open-Label, Phase 3 Study. Neurology and Therapy. 2023;12(6):1937-1958. 

  9. Jensen I, Heine J, Ruf VC, et al. Impact of Magnetic Resonance Imaging Markers on the Diagnostic Performance of the International Parkinson and Movement Disorder Society Multiple System Atrophy Criteria. Movement Disorders. 2024;39(9):1514-1522. 

  10. Duga V, Giossi R, Romito LM, et al. Long-Term Globus Pallidus Internus Deep Brain Stimulation in Pediatric Non-Degenerative Dystonia: A Cohort Study and a Meta-Analysis. Movement disorders : official journal of the Movement Disorder Society. 2024;39(7):1131-1144. 

  11. Vogt LM, Yan H, Santyr B, et al. Deep Brain Stimulation for Refractory Status Dystonicus in Children: Multicenter Case Series and Systematic Review. Ann Neurol. 2023. 

  12. Li Y-L, Lin J, Huang X, et al. Heterozygous Variants in Cause Paroxysmal Kinesigenic Dyskinesia Via Haploinsufficiency. Annals of Neurology. 2024;96(4):758-773. 

  13. Wirth T, Roze E, Delvallée C, et al. Rare Missense Variants in Are Associated with Paroxysmal Kinesigenic Dyskinesia. Movement Disorders. 2024;39(5):897-905. 

  14. Wilke C, Pellerin D, Mengel D, et al. GAA-FGF14 ataxia (SCA27B): phenotypic profile, natural history progression and 4-aminopyridine treatment response. Brain. 2023;146(10):4144-4157. 

  15. Abou Chaar W, Eranki AN, Stevens HA, et al. Clinical, Radiological and Pathological Features of a Large American Cohort of Spinocerebellar Ataxia (SCA27B). Annals of Neurology. 2024;n/a(n/a). 

 

 

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