First report of Rhinocladiella mackenziei osteomyelitis with CNS dissemination in a pediatric CGD patient post-HSCT: A case report and literature review
Abstract
Background: Rhinocladiella mackenziei is a rare neurotropic dematiaceous fungus that causes cerebral phaeohyphomycosis, predominantly in immunocompetent adults with a geographical predilection to the Middle East. Pediatric infection is exceptionally rare, and to our knowledge, no cases of combined osseous and central nervous system involvement following hematopoietic stem cell transplantation (HSCT) have been reported.
Case: We report a male child with autosomal recessive chronic granulomatous disease (CGD) who developed traumatic forearm osteomyelitis due to R. mackenziei several months after haploidentical HSCT. Subsequent neuroimaging demonstrated intracerebral abscesses despite immune reconstitution. The infection persisted despite prolonged combination antifungal therapy with voriconazole and amphotericin B and ultimately required neurosurgical excision. The patient recovered without neurological sequelae and continues on long-term posaconazole therapy.
Conclusion: This case highlights R. mackenziei as a cause of severe, invasive, disseminated fungal disease in pediatric post-bone marrow transplant patients and demonstrates extracerebral inoculation with subsequent CNS dissemination. It also underscores the importance of early recognition and a combined medical-surgical approach in management of refractory disease.
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