Abstract We report clinical and MRI findings of 17 patients with oromandibular dystonia (OMD) due to Japanese encephalitis (14) and nonspecific encephalitis (3). Their median age was 14(2–53) years and 9 were females. 8 patients had jaw open and 9 jaw close OMD. The severity ranged between 2 and 4 on a 0–4 scale, 11 patients were anarthric and needed tube feeding. Cranial MRI was abnormal in 13 patients; the abnormalities were in thalamus in 9, substantia nigra in 10, caudate in 3, globus pallidus and putamen in 2 each and pons in 1 patient. SPECT revealed hypoperfusion in thalamus in 4, basal ganglia in 1, frontal in 6, parietal in 3 and temporal in 1 patient. By 6 months, OMD regressed completely in 6, by 1 grade in 2 and remained unchanged in 7 patients. OMD in encephalitis is mainly due to JE and half of these patients improve.