Sudden cessation of baclofen can produce a withdrawal syndrome even if it was previously orally administered. We present the case of a man who exhibited signs of baclofen withdrawal syndrome during palliative sedation. Attempts were made to induce muscle relaxation with ever-increasing doses of benzodiazepine. Ultimately, control over the withdrawal syndrome was regained by using a continuous subcutaneous infusion (CSCI) of dexmedetomidine, a highly selective α2 adrenergic agonist. Very limited published reports concerning CSCI of dexmedetomidine exist. To our knowledge, this is the first case to report its use as an adjunctive agent to treat baclofen withdrawal syndrome through the subcutaneous route in the palliative care setting.