PMID- 37723545 OWN - NLM STAT- MEDLINE DCOM- 20230920 LR - 20230922 IS - 1472-6823 (Electronic) IS - 1472-6823 (Linking) VI - 23 IP - 1 DP - 2023 Sep 18 TI - Unexpected exacerbation of cryptococcal meningitis after unilateral adrenalectomy in a PMAH patient: a case report and literature review. PG - 199 LID - 10.1186/s12902-023-01457-5 [doi] LID - 199 AB - BACKGROUND: Primary 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 PRESENTATION: A 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. CONCLUSION: Cushing'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. CI - (c) 2023. BioMed Central Ltd., part of Springer Nature. FAU - Tan, Lu AU - Tan L AD - Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, Chengdu, Sichuan, 610041, P. R. China. FAU - Zhao, Lianling AU - Zhao L AD - Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, Chengdu, Sichuan, 610041, P. R. China. FAU - Tang, Guangmin AU - Tang G AD - Center of Infectious Diseases, West China Hospital, Sichuan University, Chengdu, Sichuan, 610041, P. R. China. FAU - Ren, Yan AU - Ren Y AD - Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, Chengdu, Sichuan, 610041, P. R. China. FAU - Tian, Haoming AU - Tian H AD - Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, Chengdu, Sichuan, 610041, P. R. China. FAU - Chen, Tao AU - Chen T AUID- ORCID: 0000-0002-8371-779X AD - Department of Endocrinology and Metabolism, Adrenal Center, West China Hospital of Sichuan University, 37 GuoXue Lane, Chengdu, Sichuan, 610041, P. R. China. chentao@scu.edu.cn. LA - eng GR - 2017FZ0062/Sichuan Province Science and Technology Support Program/ GR - ZYGD18022/West China Hospital, Sichuan University/ PT - Case Reports PT - Journal Article PT - Review DEP - 20230918 PL - England TA - BMC Endocr Disord JT - BMC endocrine disorders JID - 101088676 SB - IM MH - Male MH - Humans MH - Aged MH - *Meningitis, Cryptococcal/diagnosis/etiology MH - Adrenalectomy/adverse effects MH - *Cushing Syndrome/etiology/surgery MH - *Cryptococcosis MH - Adrenal Glands PMC - PMC10506208 OTO - NOTNLM OT - Case report OT - Cryptococcal meningitis OT - Cushing's syndrome OT - Immune reconstitution inflammatory syndrome OT - Primary bilateral macronodular adrenal hyperplasia COIS- The authors declare that they have no competing interests. EDAT- 2023/09/19 00:43 MHDA- 2023/09/20 06:42 PMCR- 2023/09/18 CRDT- 2023/09/18 23:51 PHST- 2022/10/18 00:00 [received] PHST- 2023/09/13 00:00 [accepted] PHST- 2023/09/20 06:42 [medline] PHST- 2023/09/19 00:43 [pubmed] PHST- 2023/09/18 23:51 [entrez] PHST- 2023/09/18 00:00 [pmc-release] AID - 10.1186/s12902-023-01457-5 [pii] AID - 1457 [pii] AID - 10.1186/s12902-023-01457-5 [doi] PST - epublish SO - BMC Endocr Disord. 2023 Sep 18;23(1):199. doi: 10.1186/s12902-023-01457-5.