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A 55-year-old HIV-positive woman presented with 16 days of fever, malaise and 10 days of oral mucocutaneous lesions, and a left supraclavicular swelling. She had no history of recent travel, exposure to birds, animals or individuals with tuberculosis. She was married and monogamous with good adherence to the highly active antiretroviral therapy (HAART), atazanavir, ritonavir and emtricitabine/tenofovir. She had a smoking history of 25 pack years of tobacco. Her husband’s HIV status was negative. The physical examination revealed normal vital signs except for a body temperature at 38.9°C. The oral examination was significant for a diffused, firm, mildly erythematous and tender mass located in the back of the right inner upper gum and hard palate. There were also lip swelling, oral candidiasis and a greenish purulent secretion in the affected gum (figure 1A). Her neck examination showed a 3×2.5 cm visible and palpable left supraclavicular lymphadenopathy (figure 1B). The rest of the physical examination was normal.
Laboratory studies revealed high levels of C reactive protein (CRP) at 20.30 mg/dL (reference value: 0.0–1.0 mg/dL), erythrocyte sedimentation rate (ESR) at 77 mm/hour (reference value: 0.0–15 mm/hour), alkaline phosphatase (ALP) at 510 U/L (reference value: 40–160 IU/L) and lactic acid dehydrogenase (LDH) at 480 U/L (reference value: 45–90 IU/L). The rest of medical investigation reports are shown in table 1.
A biopsy of the gum and hard palate mass revealed narrow budding fungal bodies with no evidence of malignancy. The papanicolaou stain enhanced the characteristic mucopolysaccharide capsule of the microorganism confirmed as Cryptococcus neoformans by Gomori methenamine silver stain (figure 2A–D). The clinical, epidemiological and histopathological findings were consistent with oral cryptococcosis. Fluconazole 400 mg intravenous daily was administrated for 2 weeks and continued for 14 weeks orally. The patient experienced complete resolution of fever, oral candidiasis, supraclavicular lymphadenopathy, gum and hard palate lesion, and normalisation of CRP, ESR, ALP and LDH over 2 weeks and is receiving outpatient follow-up and further care.
Oral and mucocutaneous cryptococcal infections have rarely been documented in the literature. A review of the literature revealed seven cases of cryptococcal infection with oral manifestations prior to 2016: five of them occurred in HIV positive and/or patients with AIDS, one in an immunosuppressed stem cell transplant recipient, and one in an immunocompetent host. The lesions were located in the palate (two cases), tongue (two cases), maxilla (one case), mandibula (one case) and salivary glands (one case).1
Cryptococcal infection can affect the lungs and almost any other organ. However, on dissemination, the fungus has an especial tropism for the central nervous system, frequently causing meningitis.1–3 Cryptococcal meningitis occurs when the CD4 count decline below 100 cells/mm3. In 2006, over a million cases of cryptococcal meningitis were reported worldwide.2 Systemic cryptococcosis currently kills over half a million individuals annually, mainly in Africa.2
Our patient is currently well controlled on HAART with an undetectable HIV viral load and a CD4 count greater than 400. To the best of our knowledge, oral cryptococcosis in an HIV-positive woman with suppressed viral load and CD4 count greater than 400 is a rare manifestation. This is probably the first case being reported.
Cryptococcus neoformans is a ubiquitous yeast discovered in 1894 by Abraham Buschke and Otto Busse who isolated it from a lesion located in the tibia of a woman.
Oral cryptococcosis is a mycosis of the oral cavity that has rarely been reported except in patients with HIV, where the use of highly active antiretroviral therapy and fluconazole can prevent and treat most cases.
Chronic, bizarre oral or maxillary pathology should indicate a systemic mycosis work up because of the risk for systemic cryptococcosis and meningitis which kills over half a million individuals annually, mainly in Africa.
We thank the Pathology Department for the histopathological diagnosis and images. We also thank the patient for allowing us to publish her clinical case for medical education purposes.
Contributors AJR conceived and drafted the first manuscript. AJR, KR, SG and SA worked on the acquisition of data, and its interpretation, and contributed to subsequent revisions of the manuscript. All authors have seen and agree with the final version.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.