Intraosseous maxillary phaeohyphomycosis mimicking an odontogenic cyst in an immunocompetent patient: a case report
Intraosseous maxillary phaeohyphomycosis mimicking an odontogenic cyst in an immunocompetent patient: a case report
DOI:
https://doi.org/10.51473/rcmos.v1i1.2026.2127Keywords:
Phaeohyphomycosis. Melanized fungi. Maxilla. Intraosseous lesion. Oral fungal infections.Abstract
Phaeohyphomycosis is a rare fungal infection caused by melanized fungi and may mimic odontogenic lesions when affecting the maxillofacial bones. Intraosseous involvement of the maxilla is exceptional, particularly in immunocompetent patients. A 50-year-old immunocompetent man presented with pain in the maxillary incisors and progressive buccal and palatal swelling. Computed tomography revealed an extensive hypodense intraosseous lesion involving teeth 22 to 12, with cortical bone destruction and extension into the nasal cavity, initially suggestive of an aggressive odontogenic cyst. Histopathological examination showed granulomatous inflammation with granular histiocytes, multinucleated giant cells, and cholesterol crystals. Grocott, PAS/PAS-D and Fontana-Masson stains demonstrated pigmented septate hyphae and spherical fungal structures consistent with dematiaceous fungi, confirming intraosseous phaeohyphomycosis. Surgical debridement associated with systemic itraconazole therapy resulted in complete resolution, with no recurrence after two years of follow-up. This case highlights the importance of including phaeohyphomycosis in the differential diagnosis of atypical intraosseous maxillary lesions and reinforces the decisive role of histopathological examination with special stains.
Downloads
References
ANDRADE, S. L.; FALEIROS, A. C.; LOPES, C. A. et al. Oral phaeohyphomycosis in a patient with squamous cell carcinoma of the lip. Brazilian Journal of Microbiology, v. 48, n. 3, p. 522–525, 2017.
ARIBANDI, M.; MCCOY, V. A.; BAZAN, C. Imaging features of invasive and noninvasive fungal sinusitis: a review. Radiographics, v. 27, n. 5, p. 1283–1296, 2007.
BONIFAZ, A.; MARTÍNEZ-SOTO, E.; CARRASCO-GERARD, E.; PENICHE, J. Subcutaneous phaeohyphomycosis: clinical and mycologic study of 10 cases. International Journal of Dermatology, v. 40, n. 2, p. 116–120, 2001.
CARDOSO, S. V.; CAMPOLINA, S. S.; GUIMARÃES, A. L. S. et al. Oral phaeohyphomycosis: case report and review of the literature. Journal of Clinical Pathology, v. 60, n. 1, p. 98–100, 2007.
CHOWDHARY, A.; MEIS, J. F.; GUARRO, J. et al. ESCMID and ECMM joint clinical guidelines for the diagnosis and management of systemic phaeohyphomycosis: diseases caused by black fungi. Clinical Microbiology and Infection, v. 20, supl. 3, p. 47–75, 2014.
GAVITO-HIGUERA, J.; MULLINS, C. B.; RAMOS-DURAN, L. et al. Sinonasal fungal infections and complications: a pictorial review. Journal of Clinical Imaging Science, v. 6, p. 23, 2016.
HE, Y.; ZHENG, H. L.; MEI, H. et al. Phaeohyphomycosis in China. Frontiers in Cellular and Infection Microbiology, v. 12, p. 895329, 2022.
QUEIROZ-TELLES, F.; DE HOOG, S.; SANTOS, D. W. C. L. et al. Phaeohyphomycosis. Clinical Microbiology Reviews, v. 30, n. 3, p. e00076-16, 2017.
RAI, A. et al. A rare case of maxillary osteomyelitis caused by Curvularia species. Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology, v. 117, n. 3, p. e231–e235, 2014.
RAWAL, Y. B.; KALMAR, J. R. Intraoral phaeohyphomycosis. Head and Neck Pathology, v. 6, n. 4, p. 481–485, 2012.
REVANKAR, S. G.; SUTTON, D. A. Melanized fungi in human disease. Clinical Microbiology Reviews, v. 23, n. 4, p. 884–928, 2010.
SÁNCHEZ-CÁRDENAS, C. D.; ISA-PIMENTEL, M.; ARENAS, R. Phaeohyphomycosis: a review. Microbiology Research, v. 14, n. 4, p. 1751–1763, 2023.
SHUKLA, M. et al. Phaeohyphomycosis masquerading as a palatal neoplasm in an immunocompetent woman. Indian Journal of Pathology and Microbiology, v. 52, n. 3, p. 396–398, 2009.
SIGAMANI, K.; NANDINI, M. S.; NARAYANAN, R. S.; RAJESH, E. Clinicopathological characteristics of fungal infections with special emphasis on dematiaceous fungi: a cross-sectional study. Bangladesh Journal of Medical Science, v. 24, supl. 10, p. 144–148, 2025.
SURESH, A. et al. COVID-19-associated fungal osteomyelitis of jaws and sinuses: an emerging clinical entity. Journal of Oral and Maxillofacial Pathology, v. 26, n. 1, p. 78–85, 2022.
YADAV, P. et al. Cytomorphologic diagnosis of phaeohyphomycosis: role of fine needle aspiration cytology. Diagnostic Cytopathology, v. 51, n. 1, p. E18–E23, 2023.
Downloads
Published
Issue
Section
Categories
License
Copyright (c) 2026 CLAUDIO PEREIRA, Patricia Freire Gasparetto, Leonardo Araújo de Andrade, Tessa de Lucena Botelho (Autor)
This work is licensed under a Creative Commons Attribution 4.0 International License.
This work is licensed under the Creative Commons Attribution 4.0 International License (CC BY 4.0). This means you are free to:
- Share — copy and redistribute the material in any medium or format.
- Adapt — remix, transform, and build upon the material for any purpose, even commercially.
The use of this material is conditioned upon proper attribution to the original author(s), providing a link to the license, and indicating if changes were made. The license does not require permission from the author or the publisher as long as these conditions are met.
The Creative Commons license logo is displayed permanently in the footer of the journal.
The copyright of the manuscript can be retained by the authors without restrictions and can be requested at any time, even after publication in the journal.
How to Cite
