BACKGROUND: Diabetic striatopathy (DS), marked by hemichorea-hemiballismus syndrome (HCHB) linked to hyperglycemia, presents as a rare complication of diabetes mellitus. CASE DESCRIPTION: This paper details a case study involving a 60-year-old woman with poorly regulated diabetes, outlining the evolution, clinical signs, imaging characteristics, and therapeutic interventions for DS. The patient exhibited persistent involuntary movements in her left upper and lower limbs, exacerbated by voluntary actions but relieved during sleep. Clinical evaluation revealed hyperkinetic movements, disrupted gait, and distinct imaging results. Treatment encompassed glycemic control and medication, resulting in notable improvement. Discussion focuses on the pathophysiology, differential diagnoses, and treatment strategies, underlining the importance of glycemic management. CONCLUSION: This study contributes to the comprehension and management of DS, emphasizing the significance of prompt identification and effective treatment approaches.