Oral paracoccidioidomycosis

Oral paracoccidioidomycosis A study of 36 South American patients Maria Regina Sposto, PhD,a Crispian Scully, PhD, MD, MDS, FDSRCFS, FDSRCS, FFD, FRCPath,h Oslei Paes de Almeida, PhD,’ Jacks Jorge, MSC,~ Edgard Graner MSc,’ and Laurence Bozzo, PhD,C Araraquara and Piracicaba, Sao Paulo, Brazil, and Bristol, United Kingdom UNESP,

UNICAMP,

AND

BRISTOL

DENTAL

HOSPITAL

AND

SCHOOLS

Paracoccidioidomycosis (South American blastomycosis) is an uncommon, progressive systemic mycosis, virtually only seen in persons who have visited Latin America. Reports of oral lesions are extremely rare in the English-language literature. Thirty-six adults with oral lesions as the first sign of paracoccidioidomycosis are described; this appears to be the largest series in the dental literature. All had chronic proliferative mulberry-like ulcerated oral lesions; the diagnosis was confirmed histologically. The gingiva or alveolar process was the typical site, but lesions were also seen particularly on the palate and lip. Most of the patients proved to have detectable pulmonary involvement. Patients with lesions in the oropharynx, tongue, or floor of mouth all had confirmed pulmonary lesions. (ORAL SI~RC. ORAL MED

ORAL PATHOI. 1993;75:461-5)

T

he systemic mycosesare a group of potentially serious and often lethal infections seen mainly in personsin the developing world or in personswho are immunocompromised. The diagnosis and management of oral lesionsof these mycoses are of increasing importance because of the growing numbers of personswho are immunocompromised.‘, ’ Paracoccidioidomycosis (South American blasto,mycosis) is found in Colombia, Venezuela, Uruguay, Argentina, and particularly Brazil, where it can be endemic in certain areas3 especially the states of Sao Paula, Rio de Janeiro, and Minas Gerais. Like other mycoses, it also may be seenin immunocompromised patients.4 However, it is possible for casesof South American blastomycosis to be found almost any-

“Department of Oral Diagnosis, Faculty of Odontology, UNESP, Araraquara. bCentre for the Study of Oral Disease, University Department of Oral Medicine. Pathology, and Microbiology, Bristol Dental Hospital and School. ‘Department of Oral Pathology, Faculty of Odontology, UNLCAMP, Piracicaba. Copyright ” 1993 by Mosby-Year Book, Inc. 0030-4220/93/$1 .OO + .I0 7/13/43088

where.’ A history of travel in South or Central America may help to establish a diagnosis.6 Oral lesionshave been recorded only from patients who have lived in or visited Brazil, Paraguay, or Venezuela,7-13but these have been single casereports in all instancesexcept our report of three patients.‘3 The first reports of paracoccidioidomycosis, however, were early this century in two patients with oral mucosal lesions and cervical lymphadenopathy.’ The disease hassincebeen reported more frequently and may have increased in prevalence.I4 The organisms appear to originate from soil and gain entry to the body mainly through the aerodigestive tract, especially through the lungs, although direct mucosal infection may be possible.‘5 Somewhat surprisingly there have not been many reports of paracoccidioidomycosis in personsinfected with the human immunodeficiency virus (HIV).‘6-20 Nevertheless, in view of the prevalence of HIV infection in South America, it is possiblethat oral lesions of paracoccidioidomycosis will soon appear in HIV disease:indeed, submandibular lymph node paracoccidioidomycosis has already been recorded in HIV patients in Brazil.20 We have therefore studied a group of 36 Brazilian 461

non-HIV-infected patients with oral lesions of paracoccidioidomycosis. PATIENTS

AND METHODS

Table I showscharacteristics of the study group ol 36 adults who presented with oral lesionsthat were confirmed histologically to be paracoccidioidomycosis.‘. ” All patient.\ M’ere South Americans. seen in Brazil in the Departments of Oral Diagnosis cxxn(UNESP) or Oral Pathology (UNICAMP), ined clinically. with oral lesion biopsies performed. Chest radiography was carried out to reveal pulmonary lesions.

.A11 paticnth Mere subsequently treated at these clinics. or bq ph>‘sicians at local hospitals or the Medical School. using systemic ketoconu/ole. or a)trimoua~olc .intl amphotericin as previously discussed.’ ” RESULTS

Most patients were adult males( XY’C.), middle aged and above (range 25 to 67 years: median 46 years). mostly white (,8I %). and in low-income occupations that involve L\ork outside (75%‘). often in the construction indus[ry. These patients were often city dwellers who irorked as bricklayers. but most had

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Fig. 2. Lesions on maxillary

463

gingiva and alveolar process.

Fig. 3. Lesions on mandibular previously been farm workers or laborers from rural areas. Most patients (83%) regularly used tobacco, but only 42% used alcohol. By definition, all had oral lesions.Oral lesionswere all painful chronic proliferative or ulcerative lesions with a characteristic mulberry-like appearance (Figs. I to 5). Many had multiple oral lesions(69%), and in those with a single lesion, it was invariably found on the alveolar process(Fig. 1) or gingiva (Figs. 2 and 3). Indeed, this was the site most commonly involved (78%), although the palate was also commonly affected (47%) (Fig. 4). Lesions were less frequently found on the lip (36%) (Fig. 5) or buccal mucosa

et al.

gingiva.

(250/o),and uncommonly in the oropharynx ( I 1%) or on the tongue or floor of mouth (1 I %). Pulmonary involvement was detected radiographitally in two thirds of the group (64%). All patients with oropharyngeal or tongue or floor of mouth lesions had contirmed pulmonary lesions. DISCUSSION This appears to be the largest study of orofacial lesions of paracoccidioidomycosis reported. Thirty-six Brazilian patients with oral paracoccidioidomycosis were mainly adult white malesof low income groups, who originated from rural areas and often used

Fig.

5. Lesionon lower lip extendsto comnlissure,Intf Ain

tobacco. The alveolar processor gingiva was by far the most common oral site affected: indeed, 78% had lesionsat that site. In the minority who had only a single oral lesion detected, the alveolar processwas the only site affected, which suggeststhat this was the initial site involved. In previous reports chronic painful oral ulceration has beenreported to involve the aveolar processes,gingiva, or palate.“.” Our study confirms the samepresentation and location. The ulcershad a mulberry-like (framboesiform) appearance with pinpoint hemorrhages. The palate was also affected but lessoften than the alveolar processesor gingiva, and all but one of the 17 patients with pala-

tal lesionswere tobacco smokers.The oropharynx and the tongue or floor of mouth were lesscommonly affected. but all patients had lung lesions and were smokers of tobacco. There is no evidence, however, from this study that tobacco smoking affects the clinical pattern of oral paracoccidioidomycosis: it could, for example, simply reflect socioeconomic status. Paracoccidioidomycosis is relevant becauselesions commonly involve the head and neck, typically the facial skin, nasal mucosa, or oral mucosa. Infection often remains subclinical or localized or may occasionally disseminate.I5 The oral mucosal lesions spread to cervical lymphatics to produce massively

enlarged lymph nodes, which then may discharge onto the skin. Hematogenous dissemination of paracoccidioidomycosis to abdominal lymph nodes, spleen, liver, adrenal glands, urogenital tract, bones, skin, gastrointestinal tract, or brain can result in life-threatening complications.” Although many of our patients had detectable pulmonary involvement (64%), this probably was an underestimate. Restrepo, Trujillo and Gomez”’ reported three patients who had paracoccidioidomycosis without clinically or radiographically detectable lung involvement, but they were able to isolate the fungus from sputum, which indicates the existence of latent lung infection. indeed, chronic pulmonary paracoccidioidomqcosis is seen in 70% to 80% of patients with paracoccidioidomycosis and appears with a cough, dyspnea, fever, weight loss, and hemoptysis. These patients often have other lesions including oral ulcers. II 2, ?I The differential diagnosis of oral paracoccidioidomycosis involves carcinoma, tuberculosis, Wegener’s granulomatosis, lupus erythematosus, North American blastomycosis, histoplasmosis, coccidioidomycosis, sarcoidosis, syphilis, granuloma inguinale, actinomycosis, leishmaniasis, and other granulomatous disorders. Besides chest radiography, oral lesional biopsy is therefore indicated. Management has been discussed previously elsewhere but is typically performed with systemic ketoconazole.‘. ‘. I3 Crispian Trust,

Scully Royal

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