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TZERBOS, KABANI. AND BOOTH

4 1. Lack EE, Neave C. Vawter GF: Hepatoblastoma. A clinical and pathologic study of 54 cases. Am J Surg6:693,1982 42. Exelby PR. Filler RM, Grosfeld JL: Liver tumors in children in the particular reference to hepatoblastoma and hepatocarcinoma: American Academy of Pediatrics Surgical Section Survey, 1974. J Pediatr Surg 10:329. 1975 43. Lack EE. Neave C, Vawter GF: Hepatocellular carcinoma. Re-

J Oral Maxillofac 50:759-760.

view of 32 cases in childhood and adolescence. Cancer 52: 1510, 1983 44. Craig JR, Peters RL, Edmondson HA, et al: Fibrolamellar carcinema of the liver: A tumor of adolescents and young adults with distinctive clinicopathologic features. Cancer 46:372, 1980 45. Farhi DC, Shikes RH, Morari PJ. et al: Hepatocellular carcinoma in young people. Cancer 52: 1516, 1983

Surg

1992

Cryptococcosis as an Exclusive Oral Presentation FOTIOS

TZERBOS, DDS,* SADRU KABANI, AND DONALD BOOTH, DMDS

Cryptococcusneoformans, Histoplasmacapsulatum, and Cococcidioides immitis are the three fungi known to cause disseminated, life-threatening disease in patients with acquired immunodeficiency syndrome (AIDS). Cryptococcosis is one of the most common opportunistic infections found in patients with AIDS, and can result in meningitis, pneumonia, or both. Infection by C neoformans usually is limited to the lungs. These infections occur through aspiration of air-borne spores that lodge in the lungs and colonize. When the fungus is limited to the lungs, a good prognosis can be expected. However, when mucocutaneous involvement occurs, dissemination via the blood stream must be suspected, because such lesions are rarely primary in nature. Disseminated cryptococcosis is now being recognized with increased frequency as a result of better diagnostic procedures, wider use of immunosuppressive drugs, and the increased incidence of human immunodeficiency virus (HIV) infection.’ Since its original description in 198 1, HIV infection has been known to have significant oral manifestations. The most common oral manifestations of HIV infections include candidiasis, hairy leukoplakia, Kaposi’s sarcoma, and atypical periodontal disease.2 Opportu-

DMD, MS,t

nistic infections, such as cryptococcosis, lymphoma and aphthouslike ulcers, are relatively rare.3 This report discusses an unusual presentation of oral cryptococcosis without evidence of disseminated infection. Report

of Case

A 54-year-old white man was referred to the oral surgery clinic of Boston University Dental School for multiple ex-

Received from Tufts University School of Dental Medicine and Boston University School of Graduate Dentistry, Boston. * Resident, Department of Oral and Maxillofaciai Surgery. t Assistant Professor, Department of Oral Pathology and Medicine, Tufts University School of Dental Medicine. $ Professor and Chairman, Department of Oral and Maxillofacial Surgery, Boston University School of Graduate Dentistry. Address correspondence and reprint requests to Dr Kabani: Department of Oral Pathology and Medicine, Tufts University School of Dental Medicine, One Kneeland St, Boston, MA 02 1 I 1.

0 1992 American

Association

0278-2391/92/5007-0019$3.00/O

of Oral and Maxillofacial

Surgeons

FIGURE I. Indurated palatal ulcer in the region of the upper right second molar.

760

EXCLUSIVELY ORAL CRYPTOCOCCOSIS

tractions. The patient had been given a diagnosis of AIDS in 1987 following admission to evaluate progressive sensory motor neuropathy of the lower extremities. The patient had been treated for Pneumocystis curinii pneumonia twice with azathioprine. Oral examination showed multiple carious teeth and a large palatal ulcer in the region of the upper right second molar which had been present for about 1 month (Fig 1). The lesion was painful on palpation. The upper right second molar was extracted and an incisional biopsy of the lesion was performed. The biopsy was positive for the presence of organisms morphologically consistent with C neoformans (Figs 2, 3). The diagnosis was confirmed by mucicarmine staining. The patient failed to return for a follow up and could not be contacted. Three weeks later, he was admitted to the University Hospital for evaluation of progressive neuropathy affecting the upper and lower extremities. On admission, the patient had a temperature of 101.6”F, white blood cell count, 1.8 X 103/pL; hemaglobin, 8.7 gm/dL; hematocrit, 26.5%: and red blood cell count, 2.38 X 106/pL. The serum electrolyte level was within normal limits. The oral surgery department was consulted regarding the oral lesion. It had remained unchanged since the day of the biopsy. Because mucocutaneous lesions are rarely primary in nature, and considering the progressive neuropathy, a diagnosis of cryptococcosis affecting the central nervous system was considered. Neurology and infectious disease consultations were obtained. Blood culture and lumbar puncture results were negative for C neoformans. The patient was treated with ketoconazole 400 mg orally four times daily. After 10 days, the palatal lesion healed completely. The patient remained hospitalized for 1 more week, and the hospital course was uneventful except for a urinary tract infection that was treated with Bactrim (Roche Labs, Nutley, NJ). The patient was discharged in stable condition to a chronic care facility.

Discussion Cryptococcus neoformans, a ubiquitous and an encapsulated yeast, is a major fungal pathogen in men. Most infections occur through inhalation of unencapsulated or sparsely encapsulated yeast.4 The spectrum of host response to cryptococcal infection is very broad.

FIGURE 3. Photomicrograph showing budding cells and pseudohyphae (methenamine silver stain, original magnification X50).

have been reports of laboratory workers who had apparent asymptomatic infections with cryptococcus neoformans.’ In addition, there are patients who have asymptomatic colonization of their airways.5 A normal host immune response either eliminates or sequesters the organism. When a serious defect in the immune system is present, widely disseminated infection may occur. 6*7Our case is unusual in that the oral lesion was the only apparent manifestation of cryptococcosis. The laboratory evaluation results, including blood and cerebrospinal fluid cultures, were negative for the organism. One can speculate that C neoformans may have colonized the upper airway of the patient during a previous infection, but the host response against the organism was able to contain it in an asymptomatic state. When the patient became immunocompromised as a result of HIV infection, the organism became reactivated and resulted in this oral manifestation in an area close to where the yeast was colonized. There

References

RGURE 2. Photomicrograph showing presence of fungal organism within histiocytic cells (hematoxylin-eosin stain, original magnification X50).

1. Glick M, Cohen G, Cheney R, et al: Oral manifestations of disseminated Cryptococcus neoformans in a patient with acquired immune deficiency syndrome. Oral Surg Oral Med Oral Path01 64454, 1987 2. Scully C, Laskaris G, Pindborg J, et al: Oral manifestations of HIV infection and their management: More common lesions. Oral Surg Oral Med Oral Pathol 7 1:158, 199 1 3. Scully C, Laskaris G, Pindborg J, et al: Oral manifestations of HIV infection and their management II: Less common lesions. Oral Surg Oral Med Oral Path01 7 1: 167, 199 1 4. Edwards V, Sutherland M, Tyrer J: Cryptococcosis of the central nervous system: Epidemiological, clinical, and therapeutic features. J Neurol Neurosurg Psychiatry 33:415, 1970 5. Bennett J: Cryptococcal skin test antigen: Preparation variables and characterization. Infect Immun 32:373, 198 1 6. Bennington L, Haber L, Morgenstem L: Increased susceptibility to cryptococcosis following steroid therapy. Dis Chest 45:262, 1964 7. Perfect J: Cryptococcosis. Infect Dis Clin North Am 3:77, 1989

Cryptococcosis as an exclusive oral presentation.

759 TZERBOS, KABANI. AND BOOTH 4 1. Lack EE, Neave C. Vawter GF: Hepatoblastoma. A clinical and pathologic study of 54 cases. Am J Surg6:693,1982 42...
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