Hard palate perforation: an unusual finding in paracoccidioidomycosis

Citation
Lgm. Castro et al., Hard palate perforation: an unusual finding in paracoccidioidomycosis, INT J DERM, 40(4), 2001, pp. 281-283
Citations number
6
Categorie Soggetti
Dermatology
Journal title
INTERNATIONAL JOURNAL OF DERMATOLOGY
ISSN journal
00119059 → ACNP
Volume
40
Issue
4
Year of publication
2001
Pages
281 - 283
Database
ISI
SICI code
0011-9059(200104)40:4<281:HPPAUF>2.0.ZU;2-O
Abstract
A 36-year-old black man presented to his dermatologist in May 1996 complain ing of mucosal lesions in the mouth, as well as perforation of the hard pal ate. 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 clear ly 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 y east structures. Paracoccidioidomycosis (PCM) serologic reactions tested po sitive for double immunodiffusion (DI), complement fixation (CF) 1:256 and counterimmunoelectrophoresis (CIE) 1.128. Hematoxylin and eosin-stained sec tions 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 sho wed budding and blastoconidia suggestive of PCM. Lung computed tomography ( CT) exhibited findings consistent with pulmonary PCM. Diagnosis of the chro nic multifocal form of PCM with oral and pulmonary manifestations was estab lished. Drug therapy was initiated with ketoconazole (KCZ) 200 mg twice dai ly, which led to clinical cure in approximately 2 months. Serum antibody va lues rose 30 days after institution of therapy (CIE 1 :256; CF 1:512), peak ing at day 60 (CIE 1:1024; CF 1:1024). Three months later the daily dose wa s reduced to 200 mg and titers declined slowly. The diameter of the perfora tion remained unchanged (Fig. 2). The hard palate perforation was corrected with a palatoplasty 27 months after initiation of drug therapy (Fig. 3). K CZ was discontinued when serologic cure was achieved after 34 months of tre atment (DI weakly positive; CIE 1:8; CF not measurable). The patient was di scharged 46 months after the first visit.