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Chronic mucocutaneous candidiasis with secondary adrenal insufficiency: a case report

Abstract number: r2164

Bastug  A.T., Huner  M.A., Akinci  E., Erbay  A., Cevik  M.A., Bodur  H.

Objective: 

Chronic mucocutaneous candidiasis is a complex of disorders in which patients have chronic and recurrent Candida albicans infections of skin, nails and mucous membranes and several other disorders such as other infectious diseases, autoimmune disorders, endocrinopathies, thymoma, dental enamel dysplasia, vitiligo, alopecia totalis and interstitial keratitis can be seen. Chronic mucocutaneous candidiasis can be familial or sporadic and is generally seen in childhood but also there are some cases reported in adults. In our case chronic mucocutaneous candidiasis with secondary adrenal insufficiency is reported.

Case: 

A 34-year- old woman was admitted with the complaint of oral and oesophageal candidiasis. The patient was suffering from oral and oesophageal candidiasis for one year period and Candida albicans which was sensitive to Amphotericin B, intermediate sensitive to Fluconazole and resistant to itraconazole was isolated from the cultures of oral and oesophageal biopsy specimens and although proper antifungal therapy was given four times, recurrent lesions were appeared within one month after the antifungal therapy. When the patient was admitted except oropharengeal and oesophageal mucous membranes there was no other tissue involvement and from the cultures of oral plaques Candida albicans was isolated and the antifungal sensitivity was same. The patient was diagnosed with cell mediated immune deficiency in which T cell activation markers of CD69 and CD25 expressions were 20–30% (half of the control of 50–60%). This test is done with various dilutions 1/50 concentration of Candida antigen. The candidin skin test with 1/400 concentration of Candida antigen was positive at 48 hours. Anti-HIV was negative and CD4, CD8 counts were normal. Caspofungine 50 mg/day intravenously was given for 14 days as antifungal therapy and the lesions were disappeared on fifth day of the therapy. Beta glucan 10 mg/day therapy was given to support the immunity. Because endocrinopathy is seen 50% of the patients with chronic mucocutaneous candidiasis the hormone levels were examined. Cortisol level was 2.6 mg/dl and the cortisol levels increased by insulin-hypoglycaemia test and synacthen test so secondary adrenal insufficiency was confirmed and deltacortril was given 5 mg/day.

Session Details

Date: 01/08/2007
Time: 00:00-00:00
Session name: XXIst ISTH Congress
Subject:
Location: Oxford, UK
Presentation type:
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