Successful management of cryptococcosis of the bilateral adrenal glands and liver by unilateral adrenalectomy with antifungal agents: a case report

Abstract

BackgroundCryptococcus species usually affect the central nervous system and lungs in immunocompromised hosts. Although the adrenal glands can be involved in disseminated cryptococcosis, primary adrenal insufficiency caused by the fungal infection is uncommon.Case presentationWe present a case of primary adrenal insufficiency with bilateral adrenal masses and liver invasion in a 43-year-old man with mild type 2 diabetes mellitus. Cryptococcosis was diagnosed by fine-needle aspiration biopsy of the liver mass. The serum cryptococcal antigen titer was elevated to 1:256. After 6 months of antifungal therapy with fluconazole and amphotericin B, the size of the liver mass was decreased, but no significant changes were observed in the bilateral adrenal masses and the serum cryptococcal antigen titer remained elevated at 1:128. To control the cryptococcosis, a laparoscopic left adrenalectomy was performed, followed by antifungal therapy. After the unilateral adrenalectomy, the size of the remaining right adrenal mass was reduced and the serum cryptococcal antigen titer declined to 1:4.ConclusionsThis is the first report describing adrenal cryptococcosis with adrenal insufficiency and liver invasion without central nervous system involvement. Adrenal cryptococcosis should be considered in the differential diagnosis for patients with bilateral adrenal masses with primary adrenal deficiency. Unilateral adrenalectomy was quite effective in controlling the cryptococcosis in this case. Even in patients with bilateral adrenal cryptococcosis, unilateral adrenalectomy should be an option for treatment of disseminated cryptococcosis.

Disseminated Cryptococcosis with Adrenal Insufficiency and Meningitis in an Immunocompetent Individual

“…Matsuda et al reported that, despite the presence of bilateral adrenal masses in their patient, they chose to perform unilateral adrenalectomy because adhesion of the right adrenal mass to the liver was anticipated, which may have caused massive bleeding. Unilateral adrenalectomy led to remarkable decreases in the serum cryptococcal antigen titers and a reduction in the size of the remaining right adrenal mass ( 11 ). If the masses in the adrenal gland or liver start to become enlarged or the Cryptococcus antigen titer is elevated, unilateral adrenalectomy should be considered.…”

Normalization of Bilateral Adrenal Gland Enlargement after Treatment for Cryptococcosis

“…In most of the reports, the enlargement was mentioned [5–8] at the diagnosis or did not change, even after the antifungal treatment was effective [9–14]. There is only one report which shows a decrease in adrenal gland enlargement after treatment with amphotericin B [15]; however, the images obtained after treatment clearly showed adrenal gland enlargement.…”

“…The infected glands showed caseating necrosis [5, 6, 10, 13, 16] or necrotizing granuloma [6, 14], accompanied by yeast-like organism, chronic inflammation with giant cells, multinucleated histiocytes, and a few lymphocytes. Although there were no histological data from the adrenal glands in this patient, infiltration of inflammatory cells into the adrenal glands could cause the enlargement.…”

Isolated adrenal cryptococcosis, diagnosed by fine‐needle aspiration biopsy: A case report

“…The patient presented with diabetes mellitus and roentgenographic findings were suggestive of a malignant tumor . Matsuda et al described a case of adrenal cryptococcosis in a patient who also presented with adrenal insufficiency . FNA cytology established the diagnosis of cryptococcus.…”

“…Cell block sections from positive FNA cases when stained with mucicarmine show a characteristic pink staining of the capsule. Radiologic imaging and FNA of the adrenal gland is often helpful in the diagnosis of adrenal gland mass lesions . Bilateral adrenal enlargement can be caused by metastatic disease, hyperplasia, or infection and a definitive diagnosis can be readily provided using FNA …”