A case of primary vertebral osteomyelitis due to an opportunistic fungus, Pseudallescheria boydii, in a child with acute myeloid leukemia, is reported. To our knowledge this is the first such case in a child, and only the second reported case in the international literature of primary spinal osteomyelitis due to this organism. We discuss the problems presented by this case both in terms of diagnosis and management. We also discuss the role of surgery and systemic antifungal chemotherapy in the treatment of deep infections due to Pseudallescheria boydii.