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Diabetic striatopathy, a rare hyperglycemia complication, is characterized by chorea/ballism and striatal anomalies on neuroimaging, usually managed with glycemic control and haloperidol. However, practical strategies for haloperidol‐resistant cases are scarce. We describe a 76‐year‐old Japanese woman with diabetic striatopathy who initially presented with polydipsia, polyuria, and lower‐extremity weakness. Despite pronounced hyperglycemia (725 mg/dL), her blood glucose levels were reduced through saline infusion and intravenous insulin. Subsequently, she developed whole‐body ballism concomitant with striatal hyperintensity on T1‐weighted magnetic resonance imaging, which initially responded to haloperidol. Upon discontinuation of haloperidol, her symptoms relapsed and did not improve with the reintroduction of haloperidol. Dopamine transporter single photon emission computed tomography revealed diminished bilateral striatal uptake, suggesting presynaptic dopaminergic dysfunction. This finding prompted the initiation of L‐dopa, which significantly improved her symptoms. This case underlines the need to consider presynaptic dopaminergic dysfunction in diabetic striatopathy patients unresponsive to standard treatments, highlighting the effectiveness of L‐dopa in such scenarios. [ABSTRACT FROM AUTHOR]