Department of Medicine and Bioregulatory Science, Graduate School of Medical Sciences, Kyushu University, Fukuoka 812-8582, Japan.
BMC Infect Dis. 2011 Dec 14;11:340. doi: 10.1186/1471-2334-11-340.
Cryptococcus 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.
We 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.
This 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.
隐球菌属通常影响免疫功能低下宿主的中枢神经系统和肺部。虽然肾上腺可能会受到播散性隐球菌病的累及,但由真菌感染引起的原发性肾上腺功能不全并不常见。
我们报告了 1 例 43 岁轻度 2 型糖尿病男性患者,其患有双侧肾上腺肿块和肝脏侵犯的原发性肾上腺功能不全。通过肝脏肿块的细针抽吸活检诊断为隐球菌病。血清隐球菌抗原滴度升高至 1:256。经过氟康唑和两性霉素 B 6 个月的抗真菌治疗后,肝肿块的大小减小,但双侧肾上腺肿块和血清隐球菌抗原滴度无明显变化,仍保持在 1:128。为控制隐球菌病,进行了腹腔镜左肾上腺切除术,随后进行了抗真菌治疗。单侧肾上腺切除术后,剩余右肾上腺肿块的大小减小,血清隐球菌抗原滴度降至 1:4。
这是首例描述无中枢神经系统累及的双侧肾上腺肿块伴原发性肾上腺功能不全和肝脏侵犯的肾上腺隐球菌病的病例报告。对于双侧肾上腺肿块伴原发性肾上腺功能不全的患者,应考虑肾上腺隐球菌病作为鉴别诊断。在这种情况下,单侧肾上腺切除术对于控制隐球菌病非常有效。即使对于双侧肾上腺隐球菌病患者,单侧肾上腺切除术也应是治疗播散性隐球菌病的一种选择。