A case of Opsoclonus-Myoclonus occurring in a young man, in association with traditional herbal medicine consumption is presented. Clinical and laboratory investigations did not reveal any of the known aetiological associations of the Opsoclonus-Myoclonus syndrome, raising the possibility that the traditional herbal medicine may be aetiologically implicated. This report highlights the need for proper identification and documentation of the contents of common herbal remedies and their possible side effects amongst Africans.