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<i>Paecilomyces</i>/<i>Purpureocillium</i> has recently been recognized as an emerging human pathogen, causing serious infection in immunocompromised and immunocompetent patients. Several predisposing factors have been reported, including foreign body implants, previous surgery, or trauma. Treatment with antifungal drugs often fails as species-specific differences in antifungal susceptibilities are one of the management challenges. Surgical debridement with or without antifungal therapy was sufficient to cure the infection in a few reported cases. Nonetheless, the surgical approach has been found to decrease the chance of dissemination and recurrence. Here, we report the first pediatric patient with chronic osteomyelitis of the femur secondary to <i>Paecilomyces</i> species, with no predisposing risk factors. Our case was successfully treated with a combination of antifungal therapy and surgical debridement. Additionally, we describe the first extensive literature review of previously reported <i>Paecilomyces</i>/<i>Purpureocillium</i> species infections in pediatric age groups.