To describe a case of oromandibular dystonia following recreational cocaine use and highlight the diagnostic and therapeutic challenges related to potential adulteration.

Cocaine remains one of the most commonly abused stimulants in the United States, with rising use linked to higher rates of cardiovascular, psychiatric, and infectious complications. While these multisystemic effects are well established, its influence on neuromodulation and motor control is less understood. Increasing evidence suggests that chronic cocaine exposure can disrupt basal ganglia circuits by blocking dopamine reuptake, leading to dystonias, tremors, or tics. Such symptoms may be mistaken for primary or psychiatric disorders, delaying appropriate care. Prolonged cocaine use, particularly in combination with antipsychotics, may heighten susceptibility to extrapyramidal side effects through dopaminergic and glutamatergic imbalance, resulting in subcortical instability.

A 25-year-old woman presented with abnormal mouth movements after a three-day episode of heavy cocaine use. Her symptoms included sustained jaw opening, excessive salivation, and difficulty eating, which resolved during sleep. She denied any preceding prescription neuroleptic use. Trials of valbenazine and trihexyphenidyl worsened dystonic movements. While clonazepam provided relief, it was short lived. An MRI revealed temporomandibular jaw arthritis due to persistent grinding. Ultimately chemodenervation with botulinum toxin injection into the pterygoid muscles under EMG guidance led to early improvement. In this case, the symptom onset following cocaine use, in the absence of neuroleptic treatment, raises concern for inadvertent exposure to a dopamine-blocking adulterant.

This case illustrates how cocaine and potential adulterants can interact to disturb dopaminergic pathways and trigger dystonia. Clinicians should consider stimulant-induced and adulterant-related causes in new-onset movement disorders. Broader toxicology testing, increased clinical awareness, and the use of botulinum toxin for refractory cases may improve patient outcomes and diagnostic accuracy.