J Oral Maxillofac

Surg

4&6&71.1990

South American Blastomycosis the Maxilla:

of

Report of a Case STEWART K. LAZOW, DDS,* R. DAVID SELDIN, DMD,f MARSHALL P. SOLOMON, DDSS

odontally involved teeth. A 4 x 2 x 1 cm crateriform, erythematous, ulcerative, granulomatous lesion was noted extending from the maxillary left first premolar to second molar region (Fig 1). All other oral tissues appeared normal. A panoramic radiograph showed loss of alveolar bone height in the region of the lesion and opacification of the left maxillary sinus (Fig 2). No oral-antral tistula was seen, and the patient denied symptoms of sinusitis. The patient was told to use intraoral saline rinses, discontinue wearing his denture, and return in 2 weeks for follow-up and possible biopsy if there was no improvement. The patient returned 2 weeks later with minimal change in the lesion. Under local anesthesia, two incisional biopsy specimens were obtained; the underlying alveolar bone appeared intact. The differential diagnosis included traumatic ulcer, verrucous or squamous cell carcinoma, and granulomatous diseases such as tuberculosis, syphilis, sarcoid, or fungal infection. On routine histologic examination of the biopsy specimens, non-necrotizing granulomas with abundant multinucleated giant cells were noted in the subepithelial tissues (Fig 3). Zones of pseudoepitheliomatous hyperplasia throughout the overlying stratified squamous epithelium were also noted. With a special silver stain (Gomori methanamine silver), yeastlike organisms with thick capsules in multiple budding forms were shown (Fig 4). A diagnosis of South American blastomycosis was made. The patient was contacted and asked if he had ever traveled to South America. He recounted that before immigrating to the United States 17 years previously, he had lived in Venezuela for 14 years. This substantiated the diagnosis of South American blastomycosis. The patient was referred to the Infectious Disease Department of Newark Beth Israel Medical Center for workup to rule out systemic mycotic involvement. The general physical and laboratory findings were all within normal limits. The chest radiograph was read as NAPD. The PPD and VDRL tests were nonreactive. A bone scan was negative. A computed tomography scan revealed opacification of the left maxillary sinus with apparent bony destruction of the lateral inferior wall, suggesting mycosis or neoplasm (Fig 5). A decision was made to explore the sinus and obtain a biopsy of the lesion. Under general anesthesia in the operating room, the patient underwent debridement of the granulomatous ulcer and a Caldwell-Luc procedure with nasal antrostomy. A grossly thickened, hemorrhagic Schneiderian membrane and multiple polyps were excised, and cultures were taken. No further nasal or paranasal sinus pathology

A review of the literature reveals only two cases of localized intraoral South American blastomycosis reported in the United States.‘** This article describes the first such case reported in the last 20 years, its treatment, and reconstruction. Report of a Case The patient, a 59-year-old Portugese American, was referred by his general dentist to our Oral and Maxillofacial Surgery office on March 19, 1987, for evaluation of a painful ulceration of the left maxilla. The patient recounted a history of multiple uneventful maxillary extractions for advanced periodontal disease 1 year previously, and subsequent insertion of full denture 2 months later. The patient had been unable to wear his denture for the past month due to the painful ulcer and had returned twice to his dentist for adjustments. The patient was a retired factory worker who had been living in the northeastern United States for 17 years. His medical history included a recently diagnosed episode of bronchitis in 1986, left lower lobe pneumonia in 1985, and syphilis in 1970 for which he had been treated with penicillin. The patient denied prior surgery, had no allergies, and was taking theophylline for wheezing. The patient denied fever, chills, or weight loss. His social history included cigarette smoking (30 pack years) and occasional social alcohol use; the patient denied chewing tobacco or narcotic usage. Orofacial examination showed that the patient was normocephalic, with no gross facial swelling or asymmetry, no cutaneous lesions, and no palpable lymphadenopathy. Intraoral examination revealed an edentulous maxilla and partially edentulous mandible with six remaining peri* Formerly, in private practice, Teaneck and Newark, NJ; presently enrolled in Downstate Medical School; Assistant Director, Oral and Maxillofacial Surgery, Kings County Hospital, Brooklyn, NY. t In private practice, Teaneck and Newark, NJ; Clinical Assistant Professor, Oral and Maxillofacial Surgery, University of Medicine and Dentistry of New Jersey, Newark. $ Associate Professor, Department of Pathology, Downstate Medical Center, Brooklyn, NY. Address correspondence and reprint requests to Dr Lazow: 179 Cedar Ln, Teaneck, NJ 07666. Q 1990 American geons

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FIGURE 1. Clinical appearance of granulomatous ulcerative lesion of the left maxillary ridge.

was noted. Histologic examination of the surgical specimen and cultures on blood agar at 30°C confirmed the diagnosis of South American blastomycosis and ruled out a neoplasm. On the advice of the Infectious Disease Department, the patient was started on intravenous amphotericin B. After three days, his BUN and creatinine levels increased to 30 and 2.2 mg/dL, respectively, necessitating discontinuation of the drug. The patient was switched to oral ketoconazole 200 mg daily, which he tolerated well. The postoperative course was unremarkable, and the patient was discharged 1 week later. He was maintained on ketoconazole for 6 months with monthly CBC, electrolyte determinations, and an SMA 12. At the &month postoperative visit, the oral ulceration had healed completely, and there were no sinus symptoms (Fig 6). The patient was taken off the ketoconazole for 3 months without recurrence. At that time, ridge augmentation with hydroxylapatite was performed in the office under intravenous sedation to reestablish alveolar bone height to permit denture wear (Fig 7). The patient is now asymptomatic after 2 years (Fig 8).

Discussion South American blastomycosis is a chronic mycotic disease caused by Blastomyces (Paracoccidioides) brasifiensis. It presents intraorally as a painful ulcerative granuloma involving almost any oral tissue: lips, tongue, gingiva, palate, tonsils, and buccal mucosa. Lymphatic involvement is common, causing hard, painful nodes that adhere to the

FIGURE 2. height.

Panoramic radiograph shows loss of alveolar bone

FIGURE 3. Photomicrograph of oral mucosa showing nonnecrotizing granulomatous inflammation in the subepithelial tissues. (Hematoxylin-eosin stain. Original magnification. x75.)

skin and u1cerate.3 The disease is endemic in South America where the fungus lives in soil and on vegetation. Ninety per cent of cases have occurred in males, with peak incidence in the 30- to 50-year age groupa There are four clinical types of manifestation: mucocutaneous, lymphatic, visceral (lungs). and mixed. ’ There are two schools of thought regarding portal of entry of the fungus: oropharyngeal or pulmonary. It was originally believed that agricultural workers in South America contracted the disease by cleaning their teeth with small fragments of wood or leaves of plants carrying the fungus.5 Bogliolo reported that the fungus may enter through periodontal tissues and subsequently reach the lymph nodes.6 The fungus has also been shown to penetrate the oral mucosa after extraction of teeth.7 However, Mackinnon’s studies implicate the lungs as the primary portal, with hematogenous dissemi-

FIGURE 4. Gomori methanamine silver stain shows yeastlike budding organisms with thick capsules 10 to 30 pm in diameter. (Original magnification, X200.)

SOUTH AMERICAN BLASTOMYCOSIS

OF THE MAXILLA

FIGURE 7. Panoramic radiograph showing hydroxylapatite augmentation of the left maxillary ridge.

FIGURE 5. tion

CT scan of left maxillary sinus reveals opacifica-

nation to mucocutaneous sites8 Indeed, pulmonary involvement is noted in over 60% of cases.’ Of particular clinical significance is the long latent period of up to 20 years after leaving the endemic area prior to clinical manifestation of the disease.4 This necessitates the postulation of endogenous reactivation of the infection secondary to decreased immunity or local trauma. Indeed, both Salman and Sheppard,’ and Joseph, Mare and Irving’ reported the occurrence of South American blastomycosis after dental extractions in patients removed from the endemic area for over 10 years. Diagnosis of South American blastomycosis depends on the clinical presentation, microscopic detection of the fungus in tissue specimens using special stains, blood agar cultures, serological complement fixation studies, and paracoccidioidin skin tests.’ A thorough travel history is crucial, as there has been no documented transmission of disease from human to human or animal to human.’

FIGURE 6. Clinical appearance of the alveolar ridge 6 months postoperatively showing resolution of the lesion.

If untreated, systemic South American blastomycosis is potentially fatal.’ The treatment of choice is amphotericin B, an antifungal agent which must be administered intravenously over a 2- to 6-hour period under close medical supervision in the hospital. Therapy is initiated at 0.25 mglkgld and gradually increased to 1.0 mglkgld, if tolerated. Adverse reactions include fever, chills, headache, nausea, vomiting, phlebitis and renal toxicity. If the BUN and creatinine levels increase significantly, then the drug must be discontinued. Aspirin, antihistamines, antiemetics, cortisteroids, heparin, and alternateday administration all have been used to decrease adverse effects.” If the patient is unable to tolerate amphotericin B, the next choice is ketoconazole, an oral antifungal agent, 200 mg daily for 6 months. Recent studies indicate that ketoconazole may even be superior to amphotericin B due to the ease of administration (oral, outpatient), lack of toxicity, and high recovery rate from disease.” The case presented is indicative of the rare, nondisseminated, mucocutaneous form of South American blastomycosis. The prior episodes of pneumonia and bronchitis may have been misdiagnosed and are suggestive of the pulmonary portal of infection. The patient was male and had been exposed to dis-

FIGURE 8. Clinical appearance after ridge augmentation.

of the alveolar ridge 1 year

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WOLF AND LEVENTON

ease while in his 40s and living in Venezuela. The long latent period of 17 years with subsequent reactivation of infection after extractions is also consistent with previously reported cases. The lack of lymph node, pulmonary, or systemic involvement confirm the localized, nondisseminated form of infection. One can hypothesize that the maxillary sinus involvement was due to local invasion or direct communication after extraction of periodontally involved teeth. The therapeutic success of ketoconazole in this patient was enhanced by the lack of disseminated disease in an otherwise healthy male. Summary An unusual case of localized South American blastomycosis of the maxilla, occurring in the United States, is reported. Clinical presentation, diagnosis, treatment, and reconstruction are reviewed. Acknowledgment Special thanks to Dr Jeremias Murillo, Chief Epidemiologist, Newark Beth Israel Medical Center, Newark. NJ.

J Oral Maxillofac 48.71-72.

References 1. Salman L, Sheppard SM: South American blastomycosis. Oral Surg 15:671, 1962 2. Joseph EA, Mare A, Irving WR: Oral South American blastomycosis in the United States of America. Oral Surg 21:732, 1966 3. Topazian RG, Goldberg MH: Management of Infections of the Oral and Maxillofacial Region. Philadelphia. PA. Saunders, 1981, p 425 4. Murray HW, Littman ML, Roberts RB: Disseminated paracoccidioidomycosis (South American blastomycosis) in the United States. Am J Med 56:209, 1974 5. Furtado T: Infection versus disease in South American blastomycosis. Int J Dermatol 14:117, 1975 6. Bogliolo L: South American blastomycosis (Lutz disease). Arch Derm Syph 61:470, 1950 7. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983, p 390 8. Mackinnon JE: The pathogenesis of South American blastomycosis. Trans R Sot Trop Med Hyg 53:487, 1959 9. Braunwald E, Isselbacher K, Petersdorf R, et al: Harrison’s Principles of Internal Medicine (ed 1 I). New York. NY,

McGraw Hill, 1987, p 743 10. Gilman AG, Goodman LS, Rall TW, et al: The Pharmacological Basis of Therapeutics (ed 7). New York, NY, MacMillan 1985, p 1226 11. Restrepo AM, Gomez I, Cano L. et al: Treatment of paracoccidioidomycosis with ketoconazole-A three year experience. Am J Med 74:48, 1983

Surg

1990

Acute iodide-induced Enlargement Salivary Glands MICHAEL WOLF, MD,* AND GEORGE LEVENTON,

of the

MDt

Report of a Case Local and systemic reactions to iodine-containing compounds have already been described.‘** Acute enlargement of the salivary glands sometimes has been observed hours to days after the administration of the iodides.3 The routine use of iodides during intravenous (IV) pyelography may lead to the same effects. yet it has rarely been encountered.4 We report an impressive bilateral enlargement of both parotid and submandibular salivary glands leading to dyspnea.

Received from the Department of Otolaryngology, Chaim Sheba Medical Center. Tel-Hashomer. and The Sackler School of Medicine, Tel-Aviv University, Tel-Aviv, Israel. * Instructor. t Associate Clinical Professor. Address correspondence and reprint requests to Dr Wolf: Department of Otolaryngology, Chaim-Sheba Medical Center, TelHashomer, 526221 Israel. 0 1990 American Association geons 0278-2391/90/4801-0013$3.00/O

of Oral

and Maxillofacial

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A 57-year-old man with symptoms of recurrent renal colic underwent IV pyelography using the sodium methylglucamine salts of diatrizoate (Urografin, Schering). He had no history of any specific hypersensitivity or allergy. Three hours after injection he complained of an increasing enlargement and tenderness of his face and upper neck. There was also mild dyspnea. Physical examination revealed an increasing enlargement of the parotid and submandibular salivary glands on both sides (Fig 1). There was mild stridor, with increasing difficulties in breathing. However, indirect laryngoscopy revealed no edematous changes of the larynx. The laboratory profile was normal, although iodine plasma levels were not examined. Treatment with corticosteroids was initiated, after which a gradual improvement was observed over the next 48 hours.

Discussion Acute enlargement of the salivary ing iodide administration, including phy, has rarely been described.4 The the reaction is not completely clear.

glands followIV pyelogramechanism of Follis’ found,

South American blastomycosis of the maxilla: report of a case.

An unusual case of localized South American blastomycosis of the maxilla, occurring in the United States, is reported. Clinical presentation, diagnosi...
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