A 31-year-old female, diagnosed as a case of depressive illness and being managed with neuroleptic medications, who went onto develop tardive dystonia progressively worsening over a 4-year duration. After a comprehensive and meticulous evaluation of her neurological and psychological stratum, globus pallidus interna lesioning was reputed as the best course of action. As intended, staged lesioning was executed bilaterally with a trivial resolution eventually succumbing into recurrence, compelling a repeat lesioning. It was inaptly discouraging to see her crippled with the plight. Determined, not to give upon her, a way out with a baclofen therapy was proposed. A test dose with a 100mcg of baclofen with an increment up to 150mcg over a 3-day period demonstrated a promising prospect. On that account, the insertion of the baclofen pump was performed with an outstanding aftermath in her neurological endeavor. Tardive dystonia is believed to be caused by striatal dopamine receptor super-sensitivity persuaded by the dopamine-antagonizing action of antipsychotic drugs. The first line of treatment being oral agents including oral baclofen, benzodiazepines, and antispasmodics. If the patient suffers from an early-onset primary generalized dystonia, then treatment with deep brain stimulation of the globus pallidus interna is the approved and preferred treatment approach. Recurrence of the symptoms despite of multiple lesioning can be overcome by intrathecal baclofen pump infusion as stated by many research. It is not uncommon to face complications in such a procedure, but the benefits outreach the risk, which makes it a choice of treatment. The use of a continuous intrathecal baclofen pump for cases with tardive dystonia refractory to conventional therapy, it has been approved as one of the safest and capable procedures.
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