Abstract

A 63-year-old business man presented himself with a chief complaint of painful oral ulcers for 3 weeks. The patient was healthy with no underlying disease. He neither drinks nor smokes. Physical examination revealed two mucosal lesions; one over the hard palate and the other at the right tonsillar fossa. The biopsy revealed ulceration with active inflammation and presence of some multi-nucleated giant cell. There is no evidence of malignancy and vasculitis. Malignant lymphoma was ruled out after immunohistochemistry staining. CBC, blood chemistry, urine examination, renal and liver function tests were normal. VDRL was non reactive. Anti-HIV, anti-HCV and HBS Ag were negative. Autoimmune profile, ANCA, anti-MPO, anti-pr3 were all negative. The patient was admitted because of high fever and inability to eat. Further investigation showed diffuse tiny calcified pulmonary nodules at both lungs on CT scan. Because Thailand is an endemic area for tuberculous infection, miliary TB is considered. AFB staining from previous biopsy and from (3×) sputum were negative. BAL and transbronchial biopsy results showed no evidence of tuberculosis infection. After 1 week of admission, the diagnosis still could not be made. Rebiopsy at the oral lesion was performed, unfortunately with no additional information. Staining for TB and fungus were negative. PCR for TB from the tissue was also negative. Eventually this patient developed unexplained adrenal crisis which lead us to discover bilateral adrenal glands enlargement. The oral lesions spontaneously disappeared without specific treatment. After one week of corticosteroid therapy, CT guided adrenal FNA was performed which later revealed chronic granulomatous inflammation with extensive adrenal necrosis. There was the presence of yeast-like organisms, morphology compatible with Histoplasma spp. Thus the patient was hospitalized again for a course of intravenous liposomal amphotericin B, he was discharged with itraconazole 400 mg/day.

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