Mills hemiplegic variant is a progressive motor neuron disease characterized by spastic ascending or descending hemiparesis or hemiplegia with no sensory involvement. We presented a 53years old male patient with a history of present complaints was difficulty on walking, and difficulty in speech then he developed left lower limb weakness followed by left upper limb weakness for the last 6 months. Electromyography showed a neurogenic pattern with denervation with reinnervation. Then, he successfully accomplished his treatment. The biggest challenge in making a diagnosis is ruling out other conditions that may have symptoms similar to this disease entity. Keywords: Mills hemiplegic, Electromyography, Motor neuron disease

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