原发性胆汁性胆管炎-自身免疫性肝炎重叠综合征合并肺隐球菌病1例报告

张明明; 刘寰; 张冬梅; 吴东波; 唐红

doi:10.12449/JCH240825

原发性胆汁性胆管炎-自身免疫性肝炎重叠综合征合并肺隐球菌病1例报告

DOI: 10.12449/JCH240825

四川大学华西医院感染性疾病研究所，感染与肝病研究室，成都 610041

基金项目:

四川大学华西医院学科卓越发展1·3·5工程项目 (ZYGD23030)

伦理学声明：本例报告已获得患者及其家属知情同意。

利益冲突声明：本文不存在任何利益冲突。

作者贡献声明：张明明负责课题设计，资料分析；张明明、张冬梅负责撰写论文；吴东波、刘寰参与收集数据；唐红、刘寰负责拟定写作思路，指导撰写并最后定稿。

详细信息

通信作者:
唐红， htang6198@hotmail.com （ORCID： 0000-0002-9790-6225）

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出版历程
- 收稿日期: 2024-01-29
- 录用日期: 2024-03-21
- 出版日期: 2024-08-25

Primary biliary cholangitis-autoimmune hepatitis overlap syndrome comorbid with pulmonary cryptococcosis： A case report

Laboratory of Infectious and Liver Diseases，Institution of Infectious Diseases，West China Hospital of Sichuan University，Chengdu 610041，China

Research funding:

1·3·5 Project for Disciplines of Excellent, West China Hospital, Sichuan University (ZYGD23030)

More Information

Corresponding author: TANG Hong， htang6198@hotmail.com （ORCID： 0000-0002-9790-6225）

摘要

摘要: 自身免疫性肝病重叠综合征表现为患者出现一种以上的自身免疫性肝病的生化、免疫、组织学或胆管造影特征，常需联合使用免疫抑制剂治疗。肺隐球菌病是一种由新型隐球菌或格特隐球菌引起的侵袭性肺真菌病，易在免疫功能低下患者中发生。本病例为1例重叠综合征，在免疫抑制治疗过程中发现并治疗肺隐球菌病，在抗真菌治疗过程中肝功能出现异常，根据患者的肝功能情况，评估了更换抗真菌药物的可行性，综合考虑后制定了在密切监测肝功能的情况下，积极治疗新型隐球菌感染的治疗策略，避免了感染的进展。提示在启动免疫抑制治疗前，除了全面评估全身感染灶，对于可疑感染灶也必须保持监测。
- 肝炎，自身免疫性 /
- 原发性硬化性胆管炎 /
- 隐球菌属 /
- 肺疾病，真菌性
Abstract: Patients with overlap syndrome （OS） of autoimmune liver disease may present with more than one biochemical， immunological， histological or cholangiography features of autoimmune liver disease （AILD） and often require a combination of immunosuppressants for treatment. Pulmonary cryptococcosis is a type of invasive pneumomycosis caused by Cryptococcus neoformans or Cryptococcus gattii and has a relatively high incidence rate in immunocompromised patients. This case report presents a patient with OS who was found to have pulmonary cryptococcosis during immunosuppressive therapy and developed abnormal liver function during antifungal treatment. Based on the liver function of the patient， the feasibility of adjusting antifungal agents was assessed， and active treatment strategies for novel cryptococcal infection were developed under the close monitoring of liver function， which helped to avoid the progression of infection. It is suggested that before the initiation of immunosuppressive therapy， systemic foci of infection should be comprehensively evaluated， and suspicious foci of infection should be monitored continuously.
- Hepatitis， Autoimmune /
- Primary Biliary Cholangitis /
- Cryptococcus /
- Lung Diseases， Fungal

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注： a，肝脏轻度慢性炎（G2S2），并见界面性炎，淋巴及浆细胞浸润；b，肝细胞花环样结构，胆管改变。

图 1 肝活检病理结果（HE染色，×100）

Figure 1. Pathological result of hepatic puncture（HE，×100）

下载: 全尺寸图片幻灯片

注： 2022年3月30日，加用美卓乐和硫唑嘌呤后第1次复查；2022年11月15日，使用伏立康唑后第1次复查。

图 2 治疗期间肝功能指标变化

Figure 2. Changing in liver biochemical biomarkers during treatment

下载: 全尺寸图片幻灯片

注： a，免疫抑制治疗前（2022年2月）；b，复查CT发现结节进展（2022年10月）；c，伏立康唑治疗5个月后（2023年3月）；d，氟康唑治疗3个月后（2023年6月）；e，氟康唑治疗7个月后（2023年10月）；f，氟康唑治疗11个月后（2024年2月）。

图 3 患者肺部病灶CT进展

Figure 3. CT signs of progression of pulmonary infection lesions

下载: 全尺寸图片幻灯片

表 1 药敏检测报告

Table 1. Drug sensitivity test report

抗生素	结果	敏感度	方法	折点
5-氟胞嘧啶	2	WT	MIC	≤8
伏立康唑	0.06	WT	MIC	≤0.25
卡泊芬净	≥16	耐药	MIC
米卡芬净	≥16	耐药	MIC
阿尼芬净	≥16	耐药	MIC
伊曲康唑	0.12	WT	MIC	≤0.25
两性霉素B	0.5	WT	MIC	≤0.25
氟康唑	4	WT	MIC	≤8
泊沙康唑	0.12	WT	MIC	≤0.25
注：患者肺泡灌洗液培养结果为新生隐球菌，为5-氟胞嘧啶、伏立康唑、伊曲康唑、两性霉素B、氟康唑、泊沙康唑野生型菌株。WT，野生型；MIC，最小抑菌浓度。

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