Objective: To present a case of early-onset Parkinson’s disease (EOPD) in a 41-year-old male with good levodopa response but complicated by early development of peak-dose dyskinesias.

Background: EOPD (onset 21-50 years) typically shows excellent levodopa responsiveness. However, these patients are predisposed to earlier motor fluctuations and levodopa-induced dyskinesias compared to late-onset patients, though such complications usually occur after several years of treatment.

Method: A 41-year-old male presented with two-year history of progressive left-sided rigidity and bradykinesia, beginning at age 39. Initial symptoms included pulling sensations in the left thigh, followed by distal left arm stiffness and a limp. Workup included MRI brain with nigrosome-1 protocol and laboratory tests to exclude secondary causes. A detailed levodopa challenge with observation of motor response and dyskinesias was performed.

Results: Off-medication exam revealed left-sided bradykinesia and cogwheel rigidity. Mild left-sided hemiatrophy of shoulder and forearm was noted. MRI showed blurred differentiation of left substantia nigra with fragmentary nigrosome-1 visualization; right-sided architecture preserved. Wilson’s disease excluded. No response to dopamine agonists. Lower levodopa doses failed to provide significant relief, necessitating rapid dose escalation. Levodopa/carbidopa (250/25 mg TDS) achieved good symptom control. At 11:00 (3.5 hours post-dose), examination during distraction revealed atetoid movements in left hand/foot (peak-dose dyskinesias). By 14:30 (7 hours post-dose), movements resolved with recurrence of axial rigidity and plastic hypertonia in left limbs (wearing-off phenomenon).

Conclusion: This case demonstrates EOPD confirmed clinically, by levodopa responsiveness, and characteristic MRI findings. Early peak-dose dyskinesias and wearing-off phenomena occurred within months of treatment optimization. Poor response to lower levodopa doses created a therapeutic dilemma: effective symptom control required dose escalation that ultimately precipitated dyskinesias. In classical PD, such complications typically develop after several years. This case highlights EOPD susceptibility to motor fluctuations and the need for careful dose titration to balance symptom control with dyskinesia management.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/early-onset-parkinsons-disease-with-early-peak-dose-dyskinesia-a-case-report/