The aim of this study is to investigate the electromagnetic sources of epileptic activity in two patients with juvenile myoclonus epilepsy (JME). The first patient was a 22-year old female with JME diagnosis by the age of 17 years old. Her initial EEG recording showed characteristic paroxysmal generalized activity with polyspike-wave complexes. She was on remission for 9 months. The second patient was a 29-year old male with JME diagnosis by the age 18 of years old. He showed an EEG recording with generalized spike-wave complexes of 3.5-4 Hz and presented a great improvement after therapeutic treatment. The MRI examinations for both patients did not disclose any focal lesions or areas of abnormal signal intensity or enhancement by contrast media. Magnetoencephalography (MEG) was recorded with a 122-channel whole-head system, 5 years after the disease onset for the first patient and 11 years for the second patient. For the first patient dipolar sources of MEG paroxysmal activity were localised at the vermis with extension up to the occipital region, whereas, for the second patient dipolar sources of MEG paroxysmal activity were localised at the cerebellar area (vermis and hemisphere). Implication of the cerebellum in JME, as suggested by MEG data in this study, is in accordance with previous reports employing functional MRI or cerebral blood flow evaluation in JME.