Orofacial Cryptococcosis: A Challenging Clinical Report and a Systematic Analysis of the Literature

Int J Surg Pathol. 2024 Feb;32(1):165-181. doi: 10.1177/10668969231169048. Epub 2023 May 4.

Abstract

Cryptococcosis is a neglected fungal disease. The scarcity of studies on oral cryptococcosis is certainly due to rarity and/or underreporting of the disease, especially in Brazil. We describe an example of orofacial cryptococcosis affecting a 57-year-old man after heart transplantation, who presented with multiple erythematous ulcers and erosions distributed in the chin, nasal cavity, labial mucosa, hard palate, and buccal vestibule. Computed tomography revealed opacities and micronodules in the lungs. Histopathological features of the oral and pulmonary lesions were compatible with Cryptococcus spp. Amphotericin B and fluconazole were used for treatment during hospitalization and itraconazole for prolonged therapy after hospital discharge. The patient has been under follow up for 6 months without signs of disease. According to a review conducted in PubMed, Web of Science, Scopus, Embase, and LILACS for data analysis of oral cryptococcosis, 26 reports were described in the literature. Predilection for men was observed (85%), with a male:female ratio of 5.5:1. The mean age of the individuals was 49 ± 15.3 years. Oral cryptococcosis mostly presented as an ulcer (n = 17). The palate and tongue were the most affected sites (n = 9 for each). Amphotericin B was the primary therapy utilized in most patients. Seventeen (65%) individuals survived. Knowledge of the clinicodemographic aspects of oral cryptococcosis is important for clinicians in decision making and surveillance.

Keywords: Cryptococcus; cryptococcosis; fungal infection; oral cavity; oral mucosa.

Publication types

  • Case Reports
  • Systematic Review

MeSH terms

  • Adult
  • Amphotericin B / therapeutic use
  • Antifungal Agents / therapeutic use
  • Cryptococcosis* / diagnosis
  • Cryptococcosis* / drug therapy
  • Cryptococcosis* / microbiology
  • Female
  • Fluconazole / therapeutic use
  • Humans
  • Male
  • Middle Aged

Substances

  • Amphotericin B
  • Antifungal Agents
  • Fluconazole