Hashimoto's encephalopathy (HE) is an uncommon syndrome with the characteristic triad of positive antithyroid antibodies (most commonly antibodies to thyroid peroxidase), response to steroids, and clinical picture presenting either as stroke-like pattern of focal neurological deficit or slowly progressive cognitive impairment. Myoclonus or tremors, seizures, and psychosis are other associated features which can be seen in HE. Herein, we report a girl with an uncommon presentation of orthostatic axial and myoclonic jerks in bilateral lower limbs in a case of HE.