mucocutaneous manifestation of cryptococcal infection: report of a case and review of the literature

J Oral Maxillofac Surg63:1543-1549, 2005

Mucocutaneous Manifestation ofCryptococcal Infection: Report of a Case

and Review of the LiteratureMehran Mehrabi, DMD, MD,* Shahrokh Bagheri, DMD, MD,†

Michael K. Leonard, Jr, MD,‡ and

Vincent J. Perciaccante, DDS§

Cryptococcus neoformans is a fungal organism thatcommonly affects patients that are seropositive forthe human immunodeficiency virus (HIV), most nota-bly patients with advanced acquired immune defi-ciency syndrome (AIDS) and CD4 lymphocyte cellcount of less than 100 copies/mL.1-3 The incidence ofcryptococcosis in patients with AIDS in the era beforehighly active antiretroviral therapy (HAART) had beenreported in large studies to range from 6% to 12%.4-7

In a recent study, cryptococcal infection was theAIDS-defining illness in 3% and the cause of death in5% of HIV-infected patients.1

In the United States there has been a decrease inthe severity and incidence of patients presenting withcryptococcal infection associated with HIV. However,it remains a leading cause of meningitis in many Afri-can nations.8-10 The decrease in the frequency ofcryptococcal infection within the United States isattributable to the availability of HAART after 1996.11

The primary site of C. neoformans infection is usuallythe respiratory tract or the central nervous system

(CNS). The primary clinical presentation may alsoinclude cutaneous, ocular, or the genitourinary man-ifestations. This organism has been described to infectalmost any area of the body. For example, several casereports document cryptococcemia, osteomyelitis, andperitonitis.4,5,12,13 In our review of the literature weidentified only 2 articles discussing the oral manifes-tation of C. neoformans.2,14 In 1987 Lynch and Naf-tolin14 described the first reported case of cryptococ-cal infection in a patient with AIDS, and in the sameyear Glick et al2 described a palatal lesion as the initialmanifestation of cryptococcosis.

As oral health care providers, we need to be awarethat an initial oral or cutaneous cryptococcal infectionmay serve as a precursor to severe life threateningcryptococcosis. In this case report, the patient pre-sented with simultaneous oral and cutaneous lesions5 months before the development of cryptococcalmeningitis.

Report of a Case

A 37-year-old man previously diagnosed with AIDS (CD4count of 125 copies/mL 5 days before admission) presentedto the hospital complaining of a headache. In the emer-gency department the patient was hallucinating and expe-riencing waxing and waning mental status. He had beensuffering from a constant left-sided headache surroundingan ulcerated lesion of the scalp for the past week (Fig 1).Additionally, the patient had been complaining of bilaterallower extremity weakness, photophobia, and chills. A lefttemporal scalp ulceration had been present for about 5months. The week prior he had been prescribed clindamy-cin by his primary care physician.

The patient mentioned that 5 months earlier he hadexperienced an episode of sudden burning sensation cen-tered around a recently discovered area of erythema on hisleft temporal scalp. He attributed this to an insect bite andtherefore did not pursue medical treatment at that time.

The patient’s past medical history was significant forHIV/AIDS diagnosed in 1998, at which time he had a CD4lymphocyte count of 307 copies/mL and viral load of281,000 copies/mL. On admission the patient was not on

*Oral and Maxillofacial Surgery Resident, Emory University, At-

lanta, GA.

†Former Resident, Oral and Maxillofacial Surgery, Emory Univer-

sity, Atlanta, GA; Currently, Fellow in Maxillofacial Trauma and

Cosmetic Surgery, Legacy Emanuel Hospital, Portland, OR.

‡Assistant Professor of Medicine, Division of Infectious Diseases,

Department of Medicine, Emory University School of Medicine,

Atlanta, GA.

§Assistant Professor, Residency Program Director, Oral and Max-

illofacial Surgery, Emory University, Atlanta, GA.

Address correspondence and reprint requests to Dr Mehrabi:

Department of Oral and Maxillofacial Surgery, Emory University,

1365 Clifton Road, NE, Suite B2300, Atlanta, GA 30322; e-mail

[email protected]

© 2005 American Association of Oral and Maxillofacial Surgeons

0278-2391/05/6310-0023$30.00/0

doi:10.1016/j.joms.2005.06.014

MEHRABI ET AL 1543

any medications. He had no history of treatment with anti-retroviral or prophylactic HIV medications.

On physical examination, the patient was an averagebuilt, well-nourished male. He appeared lethargic and wasafebrile with normal vital signs. There was a 3.5 cm crusted,dry, black ulcer on the left side of his temporal scalp (Fig 1).Intraoral examination showed diffuse white plaques on thedorsal surface of the tongue (Fig 2), which upon rubbingrevealed a raw erythematous surface. A 2 cm round ery-thematous, friable, papillary mass was observed on thetransition area of the hard and soft palate (Fig 3).

Laboratory studies included a complete blood count andcomprehensive metabolic panel. The white blood countwas 5.7 k/mL (normal, 4.0 to 8.5), with 54% lymphocytes(normal, 20% to 53%), mildly elevated alanine aminotrans-ferase at 72 U/L (normal, 0 to 40), and aspartate aminotrans-ferase at 44 U/L (normal, 0 to 37), with normal alkalinephosphatase and bilirubin levels. Urine analysis and hepati

tis serology were negative. A chest x-ray and computerizedtomography scan of the head were unremarkable. A lumbarpuncture was performed and the results are presented inTable 1.

The cerebrospinal fluid (CSF) tested positive for the cryp-tococcal antigen with a titer greater than 1:512. Subse-quently, biopsy specimens were taken from the scalp andthe palatal ulcerations. Figures 4 and 5 are low powerhematoxylin-eosin stains of the scalp and palatal ulceration,respectively, illustrating chronic granulomatous tissuechanges with numerous inflammatory cells. Figures 6 and 7are higher power hematoxylin-eosin stained photomicro-graphs showing numerous cryptococcal organisms with aclear surrounding secondary to the thick capsule and mucincontent. The organisms are of varying sizes, which is con-sistent with cryptococcus. This is in contrast to histoplas-mosis, which is not only a smaller organism but is also moreuniform in size. The confirmatory stain, as shown in Figure8, is the mucicarmine stain illustrating mucin in red.

The patient was admitted to the hospital for intravenous(IV) amphoteracin B therapy. He subsequently had a signif-icant improvement in his mental status and headache. Al-though the oral and cutaneous lesions showed signs ofimprovement after 10 days of IV amphoteracin B, they werestill present upon discharge. He was released from thehospital on oral fluconazole with follow-up at the outpatientinfectious diseases clinic.

The ulcerations were completely healed 3 months afterthe start of antifungal therapy (Figs 9, 10). Despite completerecovery, the patient was continued on suppressive antifun-gal therapy. He was started on HAART (lamivudine, zidovu-dine, and efavirenz) 5 months after his initial cryptococcalmeningitis. The patient developed resistance to non-nucle-oside reverse transcriptase (efavirenz) after 1 year and wasswitched to a new HAART regimen (tenofovir, atazanavir,ritonavir, and didanosine). He was asymptomatic at 2 yearson follow-up.

FIGURE 1. Clinical presentation of the scalp.

Mehrabi et al. Manifestation of Cryptococcal Infection. J OralMaxillofac Surg 2005.

FIGURE 2. Pseudomembranous candidiasis.


FIGURE 3. Clinical presentation, oral.


1544 MANIFESTATION OF CRYPTOCOCCAL INFECTION

Discussion

The differential diagnosis of this oral ulcerationshould include infection, neoplasia, trauma, and auto-immune disease. The immunocompromised state asseen with HIV would support an infectious or neo-plastic etiology. Oral infections (ie, fungal and viral)and neoplastic growths, both benign and malignant(ie, oral warts or squamous cell carcinoma) should beconsidered. Traumatic ulcers usually improve afterthe initial insult. Additionally, no history of traumawas elicited in this case. Autoimmune etiology wouldbe less likely because they tend to subside with im-mune deficiency. In the presence of cryptococcalmeningitis, these ulcers are highly suspicious of asimilar etiology.

It is important to recognize that in the patientdescribed HAART was not initiated immediately upondiagnosis. There are 2 reasons for this rationale. First,the increase in the number of lymphocytes, otherwiseknown as immune reconstitution syndrome, wouldresult in an intense inflammatory response. This re-sponse would exacerbate the patient’s meningitis to

the extent of requiring immunosuppressive therapy.In a review study by Shelburne et al15 12 cases ofcryptococcal immune reconstitution were found withHAART. In 6 cases of cryptococcal meningitis withimmune reconstitution, patient status worsens in 11to 58 days as the CD4 count increased from 25-45 to100-140.15

The second reason is related to poor patient com-pliance. This can play a major role in the developmentof HIV drug resistance. This is generated via a specificmutation in the HIV viral structure targeted by thedrug. A change in HAART regimen or dosing intervalcaused by side effects or compromised compliancewould allow for selective growth of mutated strain. Itis preferable to initiate HAART in conjunction withaggressive education regarding side effects and risksof poor compliance.

C. neoformans is a yeast-forming fungus that repro-duces by budding. The different strains of C. neofor-mans have been classically divided in to 2 varietiesthat include the 5 known serotypes.16 The serotypedifferences are based on cell wall antigenic variability

FIGURE 4. Scalp. (Low power magnification; hematoxylin-eosinstain.)


FIGURE 5. Oral. (Low power magnification; hematoxylin-eosinstain.)


Table 1. CSF ANALYSIS

Tube Result Normal Range

1 29 RBC, 207 WBC (lymphocyte 98%, monocyte 2%) WBC: 0-102 Protein 29, Glucose 29 Protein (15-45), Glucose (40-70)3 Culture positive for Cryptococcus neoformans

India Ink negativeNo growth

Mehrabi et al. Manifestation of Cryptococcal Infection. J Oral Maxillofac Surg 2005.

MEHRABI ET AL 1545

and mating conditions.3,16 Cryptococcus neoformansvariety neoformans includes serotypes A, D, AD, andC. neoformans variety gatti includes serotype B andC. Serotype AD is thought to be produced from sexualinteraction and therefore genetic recombination ofserotype A and D. DNA analysis has shown consider-able variety within serotypes; therefore classificationis most frequently correlated with geographic distri-

bution. Rabbit sera have been used for serologic dif-ferentiation of the various subclasses.17 Serotypes Aand D are most commonly found in soil and pigeonfeces, and serotypes B and C are found in severalspecies of the eucalyptus tree.3,16 The organism isgenerally acquired through the respiratory route, viaan environmental source and not from direct humanexposure. Consequently, contact isolation is not nec-essary. Animal exposure, particularly occupational ex-posure with pigeons, canaries, and cockatoos in-creases the risk for seroconversion. This is most likelysecondary to the aerosolization and inhalation offeces.16

The first documented cryptococcal infection wasdescribed by Buscke in 1895.18 The first CNS mani-festation was not described until almost 20 years laterby Verse.19 In 1954, Haugen and Baker20 establishedthat the pathogenesis of cryptococcosis was similar toa Mycobacterium tuberculosis infection, which in-cludes an initial pulmonary infection that is containedwithin the lung by granulomatous inflammation. Sub-sequent to autopsy studies, histopathologic findingsshowed a granulomatous inflammation surroundingthe infective organisms. The cryptococcal cell wallproduces an antiphagocytic virulent factor, whichallows the organism to survive within macrophages.This cytotoxic ability is not caused by production ofthe toxin but instead by the continuous production ofmucin that causes the bursting of the macrophages.21

If the organism survives the immune system, a gran-ulomatous wall is formed that prevents the systemicspread of the organism. The absence of this granulo-matous reaction will result in localized symptomatic

FIGURE 6. Oral. (High power magnification; hematoxylin-eosinstain.)


FIGURE 7. Scalp. (High power magnification; hematoxylin-eosinstain.)


FIGURE 8. Scalp. (High power magnification; Mucicarmine stain.)



disease or disseminated infection as may be seen inHIV, malignancy, or other immunologic compro-mised individuals. Infection in an immune competentpatient is generally asymptomatic.

In the United States a sudden rise in the incidenceof cryptococcal infection was observed with 24 doc-umented cases in 1965 to 336 cases in 1976. This maybe attributed to the emergence of HIV/AIDS.22 Theincidence of cryptococcal infection in non-HIV pa-tients is approaching 1:100,000.23 Before HAARTtherapy, 2.9% to 23.3% of patients infected with HIVhad a concomitant cryptococcal infection. With theadvent of HAART, the incidence has decreased to1.7% and 6.6% in San Francisco and Atlanta, respec-tively.23

Clinical manifestation of cryptococcal infectionmay involve any organ system. The most commonsites of infection are the central nervous system andlungs. Other commonly involved sites are the eyes,the prostate, and the skin. Symptoms may present inpatients that are HIV positive or negative. However,in patients who are HIV positive there is a greaterchance of cryptococcemia, extra pulmonary, or CNSpresentations. In addition, the India ink test that isfrequently used to identify the organism from the CSFis more commonly positive in patients that are HIVpositive.16 It is estimated that for a positive India inktest the CSF specimens need to contain greater than103 CFU/mL.16 Organisms that are cultured from HIVpositive patients tend to have a smaller capsule andform a non-mucoid colony.24 In these patients theinitial symptoms most commonly are headache andfever (60% to 100% of patients).3 CNS manifestationinvolves both the meninges and the brain paren-

chyma resulting in meningioencephalitis (67% to85%).3 Production of a gelatinous and mucoid pseudo-cyst covering the Virchow-Robin space can result inCNS manifestations that are particularly resistant totherapy.25 Non-CNS manifestations are most com-monly cryptococcal pneumonitis (10%) and crypto-coccemia (4% to 8%) among HIV-positive patients.3

Oral cryptococcal lesions can be an important clin-ical clue, suggesting a source for disseminated infec-tion, CNS Cryptococcosis, or serve as a predictor of adeveloping aggressive process. In the case report byLynch and Naftolin,14 a 41-year-old HIV-positive male(diagnosed 6 months prior) was referred for the eval-uation of a 2-week-old palatal discoloration and apersistent ulcer of the tongue. The patient was sub-sequently diagnosed with “atypical tuberculosis.” Onphysical examination there were multiple asymptom-atic violaceous patches and plaques on the palate. Inaddition, there was a 1 cm ulcer on the lateral borderof the tongue. Incisional biopsies of the palatal andtongue lesions were consistent with the diagnosis ofKaposi’s sarcoma of the palate and a C. neoformansinfection of the tongue. The cryptococcal organismwas confirmed with mucicarmine stain. The patient’scondition rapidly deteriorated and he died 44 daysafter admission.

In a second case reported by Glick et al2 in 1987, a25-year-old man with previous diagnosis of AIDS pre-sented with symptoms of shortness of breath, dehy-dration, diarrhea, loss of appetite, fevers, sweats, ahistory of a productive cough, and bilious vomiting.Upon physical examination he had a fever of 101° F,with a slightly erythematous oropharynx and a 1.0 �

FIGURE 9. Clinical presentation of the scalp at follow-up.


FIGURE 10. Oral clinical presentation at follow-up.


MEHRABI ET AL 1547

1.5 cm painful ulcer of the hard palate of unknownduration. Subsequent evaluation identified cryptococ-cal neoformans from the CSF. The patient was treatedwith IV amphoteracin for 7 days and referred to anoutpatient clinic upon discharge from hospital. Nofurther follow-up information was provided.

In both cases, no significant clinical improvementin the ulceration was reported with standard antifun-gal therapy. It is possible that the ulcers would havehealed with HAART. We emphasize that HAARTshould be administered during an asymptomatic pe-riod because immune reconstitution during severeclinical manifestation can be life threatening.

The diagnosis of cryptococcosis can be verified byserum, CSF, or urine antigen with latex agglutinationwhich is positive in more than 95% of cases.26 Themucocutaneous biopsy may be confirmed with theperiodic acid-Schiff, mucicarmine, methenamine sil-ver, and/or the Fontana-Masson stain.14,16 India inkstain may be used to show the large cryptococcuscapsule; however, it has a high false negative result.

Few treatment guidelines exist for cryptococcalinfection in immune competent patients. The ana-tomic site of infection may also dictate the treatmentmodality. In the HIV population, non-CNS infection istreated with oral fluconazole at a dose of 200 to 400mg/day.27 For a urinary tract or cutaneous manifesta-tion 3 to 6 months of treatment may be necessary. Ifthe patient cannot tolerate fluconazole, itraconazoleat 200 to 400 mg/day for 6 to 12 months may beprescribed. The treatment for cryptococcal meningi-tis, which was considered uniformly fatal before am-photeracin, consists of IV amphoteracin (0.5 to 1mg/kg/day) for 6 to 10 weeks, or amphoteracin andfluconazole for 6 to 10 weeks or amphoteracin and5-flucytosine for 2 weeks, followed by fluconazole for10 weeks.26,27 In HIV-negative patients this therapy issufficient to eradicate the infection. In HIV-positivepatients, before the advent of HAART, the dose offluconazole was maintained at 200 mg/day for theremainder of the patient’s life because of the highrelapse rate (50% to 60%).26 The immune reconstitu-tion produced by HAART has resulted in decreasedrate of relapse (5%),27 and various studies indicatethat suppressive therapy may be stopped after 1year.28

In this article, we discussed cryptococcal infectionthat frequently affects patients who are immunocom-promised. Oral ulcerations as a presenting manifesta-tion of this condition are rare. We describe a case oforal and cutaneous cryptococcal ulceration in a pa-tient with AIDS who was initially admitted for cryp-tococcal meningitis. Prompt therapy with intravenousamphoteracin B resulted in remarkable clinical im-provement; however, the oral and cutaneous ulcer-ations remained unchanged. We reviewed the litera-

ture and the common clinical presentations ofcryptococcal infection. The presence of oral and cu-taneous cryptococcal ulceration may represent asource for more severe clinical manifestations such ascryptococcal meningitis.

References1. Bartlett JG: Causes of death in U.S. patients dying of AIDS. The

Johns Hopkins hospital 2002 guide to medical care of patientswith HIV infection. 10th ed. Philadelphia, PA, LippincottWilliams & Wilkins, 2001, p 19

2. Glick M, Cohen G, Cheney RT, et al: Oral manifestation ofdisseminated Cryptococcus neoformans in a patient with ac-quired immunodeficiency syndrome. Oral Surg Oral Med OralPathol Oral Radiol Endod 64:454, 1987

3. Polis MA, Kovacs JA: Fungal infections in patients with theacquired immune deficiency syndrome. AIDS: Biology, Diagno-sis, Treatment and Prevention. (4th ed). Philadelphia, PA, Lip-pincott-Raven Publishers, 1997, p 231-242

4. Kovacs JA, Kovacs AA, Polis M, et al: Cryptococcosis in theacquired immunodeficiency syndrome. Ann Intern Med 103:533, 1985

5. Clark RA, Greer DA, Atkinson W, et al: Spectrum of Cryptococ-cus neoformans infection in 68 patients infected with humanimmunodeficiency virus. Rev Infect Dis 12:768, 1990

6. Chuck SL, Sande MA: Infections with Cryptococcus neofor-mans in the acquired immunodeficiency syndrome. N EnglJ Med 321:794, 1989

7. Dismukes WE: Cryptococcal meningitis in patient with AIDS.J Infect Dis 157:624, 1988

8. Hakim JG, Gangaidzo IT, Heyderman RS, et al: Impact of HIVinfection on meningitis in Harare, Zimbabwe: A prospectivestudy of 406 predominantly adult patients. AIDS 14:1401, 2000

9. Clumeck N, Sonnet J, Taelman H, et al: Acquired immunodefi-ciency syndrome in African patients. N Engl J Med 310:492,1984

10. Van De Perre P, Rouvroy D, Lepage P, et al: Acquired immu-nodeficiency syndrome in Rwanda. Lancet 2:62, 1984

11. Palella FJ, Delaney K, Moorman AC, et al: Declining morbidityand mortality among patients with advanced human immuno-deficiency virus infection. N Engl J Med 338:853, 1998

12. Behrman R, Masci J: Cryptococcal skeletal Infections: Casereport and review. Rev Infect Dis 12:181, 1990

13. Yinnon AM, Solages A, Treanor J: Cryptococcal peritonitis:Report of a case developing during continuous ambulatoryperitoneal dialysis and review of the literature. Clin Infect Dis17:736, 1993

14. Lynch D, Naftolin, L: Oral Cryptococcus neoformans infectionin AIDS. Oral Surg Oral Med Oral Pathol Oral Radiol 64:449,1987

15. Shelburne SA, Hamill, RJ, Rodriguez-Barradas MC, et al: Im-mune Reconstitution Inflammatory Syndrome. Emergence of aunique syndrome during highly active antiretroviral therapy.Medicine 81:213, 2002

16. Perfect JR, Casadevall A: Cryptococcosis. Infect Dis Clin NorthAm 16:837, 2002

17. Ikeda R, Shinoda T, Fukuzawa Y, et al: Antigenic characteriza-tion of cryptococcus neoformans serotypes and its applicationto serotyping of clinical isolators. J Clin Microbiol 36:22, 1982

18. Buscke A: Ueber eine durch Cocciden hervorgerufeneKrankheit des Menschen. Dtsch Med Wochenschr 21:14, 1895

19. Verse M: Ueber einen Fall von generalisierter Blastomykosebeim Menschen. Verh Dtsch Pathol Ges 17:275, 1914

20. Haugen RK, Baker RD: Primary complex of Cryptococcus andpulmonary lymph nodes. J Clin Pathol 24:1381, 1954

21. Feldmesser M, Tucker SC, Casadevall A: Intracellular parasitismof macrophages by Cryptococcus neoformans. Trends Micro-biol 9:273, 2001

22. Kaufman L, Blumer S: Cryptococcosis: The awakening giant.Proceedings of the Fourth International Conference on theMycoses. Pan-American Health Organization 356:176, 1977


23. Hajjeh R, Conn LA, Stephens DS, et al: Cryptococcosis: Popu-lation-based multistate active surveillance and risk factors inhuman immunodeficiency virus-infected persons. J Infect Dis179:449, 1999

24. Bottone EJ, Toma M, Johansson BE, et al: Poorly encapsulatedCryptococcus neoformans from patients with AIDS: I. Prelim-inary observations. AIDS Res 2:211, 1986

25. Bos HM, Hofman PAM, Schreij G, et al: Overwhelming CNSCryptococcus in AIDS. J Neurol 57:1560, 2001

26. Zuger A, Louie E, Holzman RS, et al: Cryptococcal disease in

patients with the acquired immunodeficiency syndrome: Diag-nostic features and outcome of treatment. Ann Intern Med104:234, 1986

27. Saag MS, Graybill RJ, Larsen RA, et al: Practice guidelines forthe management of Cryptococcal disease. Clin Infect Dis 30:710, 2000

28. Martinez E, Garcia-Viejo MA, Marcos MA: Discontinuation ofsecondary prophylaxis for cryptococcal meningitis in HIV-in-fected patients responding to highly active antiretroviral ther-apy. AIDS 14:2615, 2000

J Oral Maxillofac Surg63:1549-1554, 2005

Cemento-Osseous Dysplasia WithAssociated Simple Bone Cysts

Farzana Mahomed, BChD, BChD (Hons),*

Mario Altini, BDS, MDent, FCP(SA) Oral Path,†

Shabnum Meer, BChD, MDent,‡ and

Hedley Coleman, BDS, BChD (Hons), MDent, FCP(SA) Oral Path§

Cemento-osseous dysplasia (COD) is a nonneoplasticprocess usually confined to the tooth-bearing areas ofthe jaws or edentulous alveolar bone.1-4 Several clin-icopathologic forms of this disease are recognized,including solitary, multiple, florid, and periapical sub-types.1-5 These lesions all share the same histologicspectrum consisting of admixtures of bone and ce-mentum-like material in a fibrous stroma, but differprimarily in their extent of jaw involvement. In addi-tion, a rare familial form of this disease has beendescribed.2,6 An unusual association of COD withsimple bone cysts (SBCs) has infrequently been re-ported.3,7-10

The aim of this article is to report 7 additional casesof COD which were associated with SBCs, because ofthe highly unusual radiographic presentations thatcan mimic other jaw lesions.

Report of Cases

CASE 1A 43-year-old black woman presented with pain in the

lower left bicuspid area of 6 months’ duration. Clinicalexamination showed the patient to be partially dentate withthe alveolar ridge in the area of the missing mandibularteeth exhibiting slight bony hard buccal expansion. Pan-oramic radiography revealed a multilocular radiolucent le-sion in the mandible extending from the angle on the left tothe ascending ramus on the right (Fig 1). The locules wereof varying size. There were several radiopaque lesions mea-suring 0.5 to l cm in diameter in the right first molar regionand in the symphyseal and parasymphyseal regions. Somewere partially surrounded by a radiolucent rim. The lesionswere present both around the apices of the mandibularincisor and premolar teeth (Fig 2) and away from the toothroots toward the lower border (Fig 1). At biopsy, the sur-geon reported “falling into” numerous empty cavities, someof which contained blood-tinged fluid. These cavities wereeither not lined or had a thin fibrous lining. Multiple biop-sies were taken. Microscopic examination of all the speci-mens showed essentially similar histologic features, withthe lesional tissue consisting of a cellular fibrous connectivetissue containing both bony trabeculae and rounded cemen-tum-like globules (Fig 3). Dense sclerotic sheets of bone andcementum were evident in some areas. The cysts wereeither not lined at all (Fig 4A) or the lining consisted of athin layer of fibrous tissue (Fig 4B). A diagnosis of floridCOD associated with multiple SBCs was made. No furthertreatment was provided and the patient has been lost tofollow-up.

CASE 2A 54-year-old black woman presented for treatment of

several painful carious molar teeth. A panoramic radiographshowed a multilocular, “soap bubble” radiolucency extend-ing from the left ascending ramus to the sigmoid notch on

Received from the Division of Oral Pathology, Department of An-

atomical Pathology, University of the Witwatersrand, South Africa.

*Registrar.

†Professor/Head.

‡Lecturer/Specialist Consultant.

§Associate Professor, Senior Specialist.

Address correspondence and reprint requests to Dr Mahomed:

Division of Oral Pathology, School of Oral Health Sciences, Private

Bag 3, WITS, 2050 South Africa; e-mail: farzana.mahomed@nhls.

ac.za

© 2005 American Association of Oral and Maxillofacial Surgeons

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MAHOMED ET AL 1549

mucocutaneous manifestation of cryptococcal infection: report of a case and review of the literature

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