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Case Commentary: Successful use of olorofim for the treatment of multi-drug-resistant Lomentospora prolificans infection in a child

Summary

A young girl contracted a dangerous drug-resistant fungal infection in her knee after an injury, which failed to respond to standard antifungal treatments. Doctors obtained special access to an experimental antifungal drug called olorofim through a compassionate use program. The child showed improvement within two weeks and was completely cured after six months of treatment with no signs of the infection returning two years later, making this the first successful case of this new drug being used in a child.

Background

Lomentospora prolificans is a multi-drug-resistant filamentous fungus causing invasive disease in both immunocompromised and immunocompetent hosts with limited treatment options. The fungus was placed on the WHO 2022 Fungal Priority Pathogens list due to its pan-resistance to clinically available antifungals.

Objective

To describe and discuss the successful use of olorofim, an investigational antifungal agent, in treating a case of L. prolificans infection in an 8-year-old immunocompetent child with tendonitis, synovitis, and osteomyelitis.

Results

The patient showed improvement within 16 days of starting olorofim (MIC 0.06 μg/mL) through compassionate use. She continued olorofim for 6 months with complete resolution and no recurrence 2 years after initial diagnosis, representing the first successful use of olorofim in a child.

Conclusion

Olorofim represents a promising therapeutic option for difficult-to-treat L. prolificans infections with demonstrated efficacy in case reports and clinical trials, though hepatic adverse events and drug interactions require careful monitoring during extended treatment.
  • Published in:Antimicrobial Agents and Chemotherapy,
  • Study Type:Case Commentary,
  • Source: PMID: 41004262, DOI: 10.1128/aac.01072-25
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