Introduction:
10% of the subacute sclerosing panencephalitis (SSPE) presents with atypical features.

Aim:
SSPE is rare chronic encephalitis caused by persistent infection with defective measles virus. Characteristic MRI findings include signal changes (T2W and FLAIR hyperintensities) in bilateral occipital and parietal regions involving both gray and white matter. Early involvement of cerebellum and brainstem is not common.

Case study:
A 17-year-old male presented with complaints of recurrent seizures, slow walking, and behavioral abnormality. Neurological examination revealed cogwheel rigidity in all four limbs. MRI of the brain revealed asymmetrical cortical and subcortical altered signal intensities (T2W and FLAIR hyperintensity and T1W hypointensity) involving temporal and occipital lobes bilaterally (left more than right) with diffuse atrophy of cerebrum and cerebellum.

Results and discussion:
Early onset extrapyramidal features, seizures without myoclonus with MRI finding of posterior predominant asymmetrical cortical and subcortical signal abnormality is uncommon in SSPE.

Conclusions:
A high index of clinical suspicion for SSPE has to be maintained in patients haling from endemic areas, unvaccinated individuals presenting with seizures, behavioral abnormality, and extrapyramidal features.