A 32-year-old healthy woman in India developed a serious fungal infection in her sinuses caused by an extremely rare fungus called Schizophyllum radiatum. Standard laboratory tests could not identify the fungus due to its unusual characteristics, so doctors used advanced DNA sequencing to confirm the diagnosis. After surgery to remove the infected material and treatment with antifungal medication, the patient fully recovered, marking this as likely the first documented case of this type of infection in India.