Background:Though rare, primary central nervous system infection by melanized fungus Cladophialophora, is highly fatal inspite of surgical and medical intervention, posing a diagnostic dilemma, resulting in diagnostic delay, which adds to the poor outcome. Cladophialophora, being strikingly neurotropic, infects both immunocompetent and immunocompromised hosts, being more common in immunocompetent males. Among immunocompromised hosts, predominantly transplant recipients are affected. A case of brain abscess by Cladophialophora as an incidental finding in renal transplant recipient from western India is presented here. Case presentation: A 30 year old male, known case of chronic kidney disease, underwent renal transplantation. During post-transplantation period developed insidious onset headache and left-sided hemiparesis. CT and MRI imaging of head revealed presence of multiple coalesced abscesses and pus aspirate on direct microscopy revealed presence of septate filamentous fungi which was confirmed by fungal culture and a presumptive identification of Cladophialophora bantiana was made based on morphological characteristics. On definitive identification by ITS sequencing, microbe was found to be Cladophialophora bantiana strain IEC-CBM06 internal transcribed spacer 1; 5.8S ribosomal RNA gene. Patient responded well to appropriate surgical intervention and systemic antifungal therapy and is on regular follow up. Discussion & Conclusion: CNS infections by this neurotropic mould, which are usually fatal, should be taken into consideration for differential diagnosis of cerebral abscess in both immunocompetent and immunocompromised patients, as they are increasingly recognized, and timely diagnosis and prompt intervention is a good predictor of survival. Molecular identification plays a vital role in providing a timely and accurate laboratory diagnosis.