Methods: A 4-year-old girl presented with epilepsy of unknown origin. Electroencephalography and diagnostic imaging identified a lesion in her left occipital lobe. The patient was surgically treated with transoccipital lesionectomy and posterior mesial temporal resection.

Results: The histopathological diagnosis was lymphoplasmacytic infiltration of the leptomeninges and the underlying brain. Postoperatively, the patient had transient hemiparesia for 3 weeks, but recovered well otherwise. At 10 months post-surgery, there was no clinical or radiological evidence of recurrence. No adjuvant therapy was needed.

Conclusion: The small number of similar cases in the literature provide little information on which to base decisions about adjuvant therapy after surgical excision. Our patient's outcome, and the results in most cases with long follow-up indicate that total excision of the lesion is satisfactory and sufficient treatment. Keywords : Central nervous system, Langerhans' cell histiocytosis, solitary lesion