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<p><strong>Background:</strong> Deep brain stimulation (DBS) for essential tremor (ET) can cause unwanted side effects.</p><p><strong>Case Report:</strong> A patient with ET underwent unilateral dual-lead thalamic DBS. He later developed parkinsonism with atypical features and was diagnosed with progressive supranuclear palsy. During presentation for a second opinion, stimulation-induced side effects were suspected. Inactivation of DBS resolved atypical features and superimposed idiopathic Parkinson Disease (PD) was diagnosed.</p><p><strong>Discussion:</strong> This case illustrates the importance of recognizing the possible influence of stimulation-induced side effects and discusses when to utilize dual-lead DBS for ET and the co-occurrence of ET and PD.</p>