Background: Syncephalastrum racemosum is a saprophytic and ubiquitous fungus found in soil, mainly in tropical and subtropical areas, usually does not cause human infections and if it does, affects immunocompromised persons. It mainly causes subcutaneous infections and onychomycosis and rarely pulmonary infections. Case description: We report a case of a 36-year-old female, diagnosed with pulmonary tuberculosis and on antitubercular treatment for 1 month, presented to the emergency with complaints of shortness of breath, fever, cough with sputum production and mildly blood-stained sputum since 3 days. The chest X-ray showed bilateral infiltrates in the lower zones. Sputum investigations revealed AFB negativity, sterile pyogenic culture, and positive S. racemosum fungal culture. Deoxycholate amphotericin B was started on the basis of sputum reports, but unfortunately, the patient expired within 1 day of starting treatment. Conclusion: The above case describes infection by a rare fungal species in an immunocompromised patient with tuberculosis. Few cases are reported, so limited data are available to understand the complete disease implications. However, we should keep it as a differential in an immunodeficient patient with an invasive infection for timely diagnosis and treatment.