Unexpected exacerbation of cryptococcal meningitis after unilateral adrenalectomy in a PMAH patient: a case report and literature review

BMC Endocrine Disorders

Case Report

Guangmin Tang¹  Tao Chen²  Lu Tan²  Yan Ren²  Haoming Tian²  Lianling Zhao² 

[1] Center of Infectious Diseases, West China Hospital, Sichuan University, 610041, Chengdu, Sichuan, P. R. China;Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, 610041, Chengdu, Sichuan, P. R. China;

关键词: Primary bilateral macronodular adrenal hyperplasia;    Cryptococcal meningitis;    Cushing’s syndrome;    Immune reconstitution inflammatory syndrome;    Case report;   

DOI  :  10.1186/s12902-023-01457-5

 received in 2022-10-18, accepted in 2023-09-13,  发布年份 2023

来源: Springer

【 摘 要 】

BackgroundPrimary bilateral macronodular adrenal hyperplasia (PMAH) combined with infection by an opportunistic pathogen is complicated. Clinical evidence on managing PMAH patients with infections by opportunistic pathogens is insufficient.Case presentationA 66-year-old male was admitted with bilateral adrenal masses and was diagnosed with PMAH. Fever and disturbance of consciousness appeared after laparoscopic left adrenalectomy. Cryptococcal meningitis was confirmed by cerebrospinal fluid (CSF) culture. The exacerbation of his medical condition was suspected to result from immune reconstitution inflammatory syndrome (IRIS), and he had been treated with antifungal therapy and glucocorticoid replacement, but he responded poorly and eventually died of multiorgan failure. We summarized the clinical observations of 12 Cushing's syndrome (CS) patients infected by Cryptococcus. Seven out of nine patients who were treated for cryptococcus infection before receiving CS survived, while three patients treated for cryptococcus infection after CS treatment developed signs of IRIS and eventually died.ConclusionCushing's syndrome, complicated with cryptococcal infection, has a high mortality rate, mainly when IRIS emerges. Carefully identifying the presence of the suspected infection, and controlling cryptococcal infection before removing the culprit adrenals could be the rational choice.

【 授权许可】

CC BY   
© BioMed Central Ltd., part of Springer Nature 2023

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