Pilomyxoid astrocytoma (PMA) is a rare variant of astrocytoma that is usually present in the hypothalamic and chiasmatic areas in the paediatric population. PMA shares many similar histopathological features to Pilocytic astrocytoma (PA), with some notable differences in its radiological and histopathological findings. On the contrary, PMA has been reported to behave more aggressively in its clinical progression than PA. Here, we describe a rare case of PMA in a 25-year-old female involving the temporal lobe, presenting with recurrent partial seizures. To our knowledge, this is the first reported case of PMA presenting in the temporal lobe in an adult female with an atypical location of the tumour, uncommon age group, and unusual radiological features being unique in this case report.