| Summary: | Abstract Background Cerebellar malformations are broadly categorized into hypoplasias and dysplasias, which are further divided based on focal or diffuse involvement. Both hypoplasias and dysplasias are usually associated with cerebral involvement or as a part of syndromes. Isolated unilateral cerebellar dysplasia is, however, extremely unusual. Case presentation We present a rare case of isolated unilateral cerebellar dysplasia, in a 25-year-old male patient, who came to the Neurology clinic with complaints of infrequent seizure episodes since childhood. Conclusion Notable isolated unilateral cerebellar dysplasia is a rare entity requiring symptomatic treatment, with an anomalous development process being the likely aetiology.
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