Hard palate perforation:: an unusual finding in paracoccidioidomycosis

被引:14
|
作者
Castro, LGM
Müller, AP
Mimura, MAM
Migliari, DA
机构
[1] Univ Sao Paulo, Sch Med, Hosp Clin, Div Dermatol, Sao Paulo, Brazil
[2] Univ Sao Paulo, Sch Dent, Hosp Clin, Div Dermatol, Sao Paulo, Brazil
关键词
D O I
10.1046/j.1365-4362.2001.01212.x
中图分类号
R75 [皮肤病学与性病学];
学科分类号
100206 ;
摘要
A 36-year-old black man presented to his dermatologist in May 1996 complaining of mucosal lesions in the mouth, as well as perforation of the hard palate. The lesions had started approximately 7 months before and had worsened gradually. Other complaints included odynophagia, dysphagia, mild dyspnea, and dry cough. The patient was in good general health, but reported a 3 kg weight loss over the previous semester. The hard and soft palate presented erythematous ulcers with a finely granulated base and irregular, but clearly defined margins. A perforation (diameter, 0.5 cm) of the hard palate was seen in the center of the ulcerated region (Fig. 1). Direct examination of 10% KOH cleared specimens showed typical double-walled, multiple budding yeast structures. Paracoccidioidomycosis (PCM) serologic reactions tested positive for double immunodiffusion (DI), complement fixation (CF) 1:256 and counterimmunoelectrophoresis (CIE) 1.128. Hematoxylin and eosin-stained sections of oral lesions showed an ulcer covered by a fibrous leukocytic crust, with a lymphoplasmacytic infiltrate, as well as multinuclear giant cells containing round bodies with a double membrane. Gomori-Grocott staining showed budding and blastoconidia suggestive of PCM. Lung computed tomography (CT) exhibited findings consistent with pulmonary PCM. Diagnosis of the chronic multifocal form of PCM with oral and pulmonary manifestations was established. Drug therapy was initiated with ketoconazole (KCZ) 200 mg twice daily, which led to clinical cure in approximately 2 months. Serum antibody values rose 30 days after institution of therapy (CIE 1 :256; CF 1:512), peaking at day 60 (CIE 1:1024; CF 1:1024). Three months later the daily dose was reduced to 200 mg and titers declined slowly. The diameter of the perforation remained unchanged (Fig. 2). The hard palate perforation was corrected with a palatoplasty 27 months after initiation of drug therapy (Fig. 3). KCZ was discontinued when serologic cure was achieved after 34 months of treatment (DI weakly positive; CIE 1:8; CF not measurable). The patient was discharged 46 months after the first visit.
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页码:281 / 283
页数:3
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