Scedosporium apiospermum complex members are opportunistic fungi that can be found inenvironments such as soil and polluted water. In this report, we aimed to present a case of mycetomacaused by Scedosporium apiospermum complex that developed in a 40-year-old female patient withimmunocompetent system and diagnosed by fungal culture. In the anamnesis of the patient whoadmitted in 2015 with the complaint of more than one fistulized discharge wound, pain and swelling inthe dorsal of the right hand and wrist; it was learned that her complaints started about 20 years ago witha slight swelling on the back of the wrist, and when it worsened, the abscess was drained and antibiotictreatment was initiated in a private surgical center. However, it was learned that she did not benefit fromthe treatments, and over time, fistulized, yellow-discharged wounds appeared on the back of her handand wrist, and she had undergone various surgical interventions and used antibiotics. Routine laboratorytests of the patient, who did not have an underlying chronic disease, were normal. Magnetic resonanceimaging (MRI) and X-ray findings were compatible with osteomyelitis and ‘dot in circle’ sign seen on MRIwas characteristic for mycetoma. Pathological examination was interpreted as active chronic inflammatoryreaction in the soft tissue and chronic osteomyelitis. Mycobacteria, bacteriological and fungal culturesof the two biopsy samples taken during surgical debridement and one month later were performed.Bacteriological and mycobacterial cultures were negative, while Scedosporium genus grew in the fungalcultures of the both samples. Isolates were identified as Scedosporium apiospermum/Pseudallescheria boydii with MALDI Biotyper (Bruker Daltonics, Bremen, Germany) system and Scedosporium boydi by sequenceanalysis of the ITS region. The antifungal susceptibility tests were performed according to CLSI M38-A2criteria, and were evaluated at the 72nd hour. The minimum inhibitory concentration (MIC) values offluconazole, caspofungin, amphotericin B, itraconazole, vorikonazole, posaconazole and isavuconazolewere > 64 µg/ml, 16 µg/ml, 4 µg/ml, 16 µg/ml, 0.25 µg/ml, 2 µg/ml and 0.25 µg/ml, respectively.Voriconazole and terbinafine treatment was initiated. In the control performed in the 9th month of thetreatment, it was observed that the complaints of discharge, pain and swelling were resolved, pain andswelling complaints were recovered, fistula tracts were closed and joint movements were painless. In thecontrol MRI performed at 15th and 18th months, it was observed that there was no soft tissue involvementand the findings were compatible with osteoarthritis after infective osteomyelitis. This case whose long term complaints improved with mycological diagnosis and appropriate treatment; reveals the importanceof keeping mycological diagnosis in mind in chronic bone and soft tissue infections. Identifying the fungusto the genus and species level and arranging the treatment according to the antifungal susceptibility testresults are very important in patient management.