Cilj: Generalizirana distonija karakterizirana je nevoljnim kontrakcijama mišića koje dovode do pojave abnormalnog držanja i ponavljajućih pokreta. Duboka stimulacija mozga (engl. Deep Brain Stimulation, DBS) učinkovita je u distoniji otpornoj na lijekove, međutim, postoji manjak radova o dugoročnom učinku. Cilj rada je prezentirati dugoročni učinak primjene DBS-a kod pacijenata s generaliziranom distonijom. Prikaz slučaja: Pacijent je 29-godišnji muškarac s generaliziranom distonijom. Nakon rođenja primijećena je faciopareza po perifernom tipu, što je bilo pripisano cerebralnoj paralizi. Prvi simptomi, problemi s pisanjem zbog grčenja ruke, primijećeni su sa sedam godina. S petnaest godina primijećeni su problemi u govoru i pogoršanje distonije ruke. Distonične kretnje i držanje postupno se pogoršavalo, uzrokujući probleme s hodanjem i narušavajući kvalitetu života pacijenta. Simptomi se nisu popravljali unatoč primjeni farmakoterapije i kada je pacijent imao dvadeset i jednu godinu implantirane su mu elektrode u bazalne ganglije, što je drastično poboljšalo distonične kretnje, držanje i hod. Nakon operacije, unatrag osam godina, pacijent je redovito praćen, elektrode su reprogramirane uz kontinuiranu fizikalnu i terapiju govora. U dva navrata nastupio je povratak kliničke slike radi ispražnjene baterije neurostimulatora koja je prvi put zamijenjena nepunjivom, a drugi put punjivom baterijom, što je dovelo do potpunog oporavka. Zaključak: Idiopatska distonija često je neprepoznata. Iako je DBS sigurna i učinkovita metoda, nije dovoljno korištena za liječenje distonije otporne na lijekove. Ovaj slučaj pokazao je perzistentan i izvrstan učinak DBS-a na generaliziranu distoniju i pacijentovu kvalitetu života kroz duži vremenski period.; Aim: Generalized dystonia is characterized by involuntary muscle contractions leading to abnormal postures and repetitive movements. Deep brain stimulation (DBS) is effective in medication-refractory dystonia, but there is a lack of studies about long-term effect. The goal of this case report is to show long-term effect in a patient with generalized dystonia treated with DBS. Case report: We present a case of a 29-years-old patient with isolated generalized dystonia. At birth, peripheral facial palsy was noticed which was attributed to cerebral paralysis. First symptoms noticed at 7-years-old were problems with handwriting due to hand spasms. At 15, speech problems started and dystonia of his hand worsened. Then, dystonic movements and postures gradually expanded causing walking problems and poor quality of life. Symptoms did not improve with available medications and at the age of 21 electrodes were implanted into basal ganglia which extremely improved his dystonic movements, posture and gait. After the surgery, during these eight years, the patient was regularly controlled, electrodes were reprogramed with continuous physical and speech therapy. Twice he presented with returned complete clinical presentation due to the empty battery of the neurostimulator that was replaced first with a non-rechargeable and then with a rechargeable battery leading to complete improvement. Conclusion: Idiopathic dystonia is often unrecognized. Although, DBS is a safe and effective method, it is not used enough for treating medication-refractory dystonia. Our case has shown persistent and excellent effect of DBS on generalized dystonia and patients’ quality of life over a long period of time.