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Article(id=1198558269032726836, tenantId=1146029695717560320, journalId=1189873630562394117, issueId=1198558265329152414, articleNumber=null, orderNo=null, doi=10.11855/j.issn.0577-7402.1209.2024.0512, pmid=null, cstr=null, oa=null, hot=null, price=null, onlineType=0, articleFormat=0, articleType=null, articleTypeStr=research-article, receivedDate=1694448000000, receivedDateStr=2023-09-12, revisedDate=null, revisedDateStr=null, acceptedDate=1697385600000, acceptedDateStr=2023-10-16, onlineDate=1763688159473, onlineDateStr=2025-11-21, pubDate=1722096000000, pubDateStr=2024-07-28, doiRegisterDate=null, doiRegisterDateStr=null, onlineIssueDate=1763688159473, onlineIssueDateStr=2025-11-21, onlineJustAcceptDate=null, onlineJustAcceptDateStr=null, onlineFirstDate=null, onlineFirstDateStr=null, sourceXml=null, magXml=null, createTime=1763688159473, creator=13701087609, updateTime=1763688159473, updator=13701087609, issue=Issue{id=1198558265329152414, tenantId=1146029695717560320, journalId=1189873630562394117, year='2024', volume='49', issue='7', pageStart='733', pageEnd='854', issueExtLink='null', onlineDate='null', pubDate='null', beforeIssueId=null, nextIssueId=null, price=null, status=1, issueComplete=1, articleOrder=1, issueType=-1, specialIssue=0, createTime=1763688158589, creator=13701087609, updateTime=1763689196450, updator=13701087609, preIssue=null, nextIssue=null, ext={EN=IssueExt(id=1198562618517581944, tenantId=1146029695717560320, journalId=1189873630562394117, issueId=1198558265329152414, language=EN, specialIssueTitle=, coverIllustrator=null, specialIssueEditor=, specialIssueAbout=), CN=IssueExt(id=1198562618517581945, tenantId=1146029695717560320, journalId=1189873630562394117, issueId=1198558265329152414, language=CN, specialIssueTitle=, coverIllustrator=null, specialIssueEditor=, specialIssueAbout=)}, issueFiles=null}, startPage=783, endPage=789, ext={EN=ArticleExt(id=1198558269305356607, articleId=1198558269032726836, tenantId=1146029695717560320, journalId=1189873630562394117, language=EN, title=IgG4-related hepatic inflammatory pseudotumor complicated with hepatic tissue infection: a case report and literature review, columnId=1190310109000602400, journalTitle=Medical Journal of Chinese People’s Liberation Army, columnName=Clinical Research, runingTitle=null, highlight=null, articleAbstract=

Objective To report the clinical and imaging characteristics of a case with the clinical manifestation of multiple lung and liver nodules, diagnosed as IgG4-related hepatic inflammatory pseudotumor (HIPT) complicated by hepatic tissue-infection, and review the literature in order to facilitate clinical diagnosis and differential identification of IgG4-related HIPT. Methods A retrospective analysis was conducted on the medical records of a patient with IgG4-related disease (IgG4-RD) characterized by multiple lung and liver nodules. A literature review was performed by searching Chinese and English databases to summarize the clinical and imaging characteristics of IgG4-related HIPT and hepatic tissue infection. Results The case involved a 64-year-old female admitted to the Rheumatology and Immunology Department of the First Medical Center of Chinese PLA General Hospital due to "poor appetite and fatigue for over a year, and dry cough for four months". She presented with multiple nodules in the lungs and liver, without involvement of the eyelids, salivary glands, submandibular glands, or pancreas. Laboratory test results revealed elevated serum IgG4 levels at 14.1 g/L and C-reactive protein (CRP) at 82.1 mg/L. Pulmonary CT scans indicated multiple solid nodules in both lungs with clear boundaries. Abdominal contrast-enhanced MRI revealed a nodule in liver segment S7 with a pseudocapsule around it, clear boundaries, and uniform enhancement; another nodule in liver segment S5 with blurred boundaries and ring enhancement. The final diagnosis of the liver nodules was confirmed by pathological and metagenomic sequencing to be an IgG4-related HIPT in segment S7 and hepatic tissue infection in segment S5. After a full course of anti-infection and treatment with methylprednisolone and leflunomide, follow-up imaging showed near-complete resolution of the lung and liver nodules. Literatures were searched in China National Knowledge Infrastructure (CNKI), Wanfang, and PubMed databases (up to September 2023), and no case of IgG4-RD complicated with both liver involvement and infection was found. A total of 26 cases of IgG4-RD involving the liver have been reported so far, predominantly in males (92.3%), with an average age of 51 years. Most patients presented with abnormal liver function as the initial symptom, with normal blood inflammatory markers. Imaging typically shows a single nodule in 88.5% of cases, with clear boundaries and uniform enhancement, as well as ring enhancement. Concurrent involvement of the pancreas and biliary tract is common. Pathology is the gold standard for confirming the disease. Conclusions This case reports coexistence of IgG4-related HIPT and infection within multiple hepatic nodules. In the diagnosis and treatment of patients with IgG4-RD presenting with multiple hepatic nodules, if the imaging characteristics of the nodules are inconsistent, it is necessary to consider the possibility of the underlying disease coexisting with other conditions, which can be easily misdiagnosed. Actively obtaining pathological tissue is crucial for aiding in the definitive diagnosis.

, correspAuthors=Zheng Zhao, authorNote=null, correspAuthorsNote=
E-mail:
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目的 报道1例以多发肺、肝结节为临床表现的IgG4相关性肝脏炎性假瘤(HIPT)合并肝组织感染患者的临床影像特点并复习文献，以期帮助临床医师诊治及鉴别IgG4相关性肝脏炎性假瘤。方法 回顾性分析1例多发肺、肝结节的IgG4相关性疾病(IgG4-RD)患者的病例资料，并通过检索中英文数据库，综合文献结果，总结IgG4相关性HIPT和肝组织感染的临床影像特点。结果 本例为64岁女性，因“纳差乏力1年余、干咳4个月”至解放军总医院第一医学中心风湿免疫科住院，仅有肺、肝多发结节，无眼睑、腮腺、颌下腺、胰腺等受累，血IgG4 (14.1 g/L)及C反应蛋白(CRP，82.1 mg/L)水平均升高。肺CT提示双肺多发实性结节、边界清楚。腹部增强磁共振检查显示肝S7段结节，周围有假包膜，边界清楚，均匀强化；肝S5段结节，边界模糊，环形强化；最终肝结节通过病理和病原学宏基因测序证实S7段结节为IgG4相关性HIPT，S5段结节为肝组织感染。经足疗程抗感染和甲泼尼龙、来氟米特治疗后，复查肺、肝结节基本消失。检索中国知网、万方数据知识服务平台、PubMed数据库(截至2023年9月)，未见IgG4-RD肝脏受累同时合并感染的病例报道。目前共报道26例IgG4-RD肝脏受累病例，男性居多(92.3%)，平均年龄51岁，多以肝功能异常为首发症状就诊，血炎症指标正常，影像表现以单个结节多见(88.5%)，边界清楚，均匀强化和环形强化均可见，常同时合并胰腺、胆道受累，病理为确诊该疾病的金标准。结论 本例肝内多发结节为IgG4相关性HIPT与感染共存。在诊治以肝多发结节为表现的IgG4-RD患者时，若结节影像学特征不一致，需考虑原发病与其他情况并存的情况，该病容易误诊，须获取病理组织帮助确诊。

, correspAuthors=赵征, authorNote=null, correspAuthorsNote=
赵征，E-mail：
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廖思敏，医学博士，主治医师，主要从事结缔组织病及关节炎等疾病的诊治方面的研究

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廖思敏，医学博士，主治医师，主要从事结缔组织病及关节炎等疾病的诊治方面的研究

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廖思敏，医学博士，主治医师，主要从事结缔组织病及关节炎等疾病的诊治方面的研究

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BMC Pulm Med, 2020, 20(1): 218., articleTitle=Lung nodules and IgG4 related disease: a single-center based experience, refAbstract=null), Reference(id=1198589377996751505, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, doi=null, pmid=null, pmcid=null, year=2015, volume=94, issue=15, pageStart=null, pageEnd=null, url=null, language=null, rfNumber=[28], rfOrder=27, authorNames=Inoue D, Yoshida K, Yoneda N, journalName=Medicine (Baltimore), refType=null, unstructuredReference=Inoue D, Yoshida K, Yoneda N, et al. IgG4-related disease: dataset of 235 consecutive patients[J]. Medicine (Baltimore), 2015, 94(15): e680., articleTitle=IgG4-related disease: dataset of 235 consecutive patients, refAbstract=null), Reference(id=1198589378143552147, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, doi=null, pmid=null, pmcid=null, year=2015, volume=67, issue=9, pageStart=2466, pageEnd=2475, url=null, language=null, rfNumber=[29], rfOrder=28, authorNames=Wallace ZS, Deshpande V, Mattoo H, journalName=Arthritis Rheumatol, refType=null, unstructuredReference=Wallace ZS, Deshpande V, Mattoo H, et al. IgG4-related disease: clinical and laboratory features in one hundred twenty-five patients[J]. Arthritis Rheumatol, 2015, 67(9): 2466-2475., articleTitle=IgG4-related disease: clinical and laboratory features in one hundred twenty-five patients, refAbstract=null), Reference(id=1198589378227438231, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, doi=null, pmid=null, pmcid=null, year=2018, volume=12, issue=5, pageStart=493, pageEnd=497, url=null, language=null, rfNumber=[30], rfOrder=29, authorNames=张霞, 张文, journalName=中华临床免疫和变态反应杂志, refType=null, unstructuredReference=张霞, 张文. IgG4相关性疾病的模拟和重叠[J]. 中华临床免疫和变态反应杂志, 2018, 12(5): 493-497., articleTitle=IgG4相关性疾病的模拟和重叠, refAbstract=null)], funds=null, companyList=[AuthorCompany(id=1198589367146086666, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, xref=null, ext=[AuthorCompanyExt(id=1198589367154475275, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, companyId=1198589367146086666, language=EN, country=null, province=null, city=null, postcode=null, companyName=null, departmentName=null, remark=Department of Rheumatology and Immunology, the First Medical Center of Chinese PLA General Hospital, Beijing 100853, China), AuthorCompanyExt(id=1198589367158669580, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, companyId=1198589367146086666, language=CN, country=null, province=null, city=null, postcode=null, companyName=null, departmentName=null, remark=解放军总医院第一医学中心风湿免疫科，北京 100853)])], figs=[ArticleFig(id=1198589371390722513, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Fig.1, caption=Immunohistochemical staining of nodule in liver segment S7 of 1 patient with IgG4-related HIPT and hepatic tissue infection (×200), figureFileSmall=a5+OgRU7AOgddxP2f7X3ig==, figureFileBig=hN/HXoVAKhTG7BCGmvd0qw==, tableContent=null), ArticleFig(id=1198589371503968730, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=图1, caption=1例IgG4相关性HIPT合并肝组织感染患者肝S7段结节免疫组化染色(×200)

HIPT. 肝脏炎性假瘤

, figureFileSmall=a5+OgRU7AOgddxP2f7X3ig==, figureFileBig=hN/HXoVAKhTG7BCGmvd0qw==, tableContent=null), ArticleFig(id=1198589371638186469, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Fig.2, caption=Comparison of lung CT scans in 1 patient with IgG4-related HIPT and hepatic tissue infection before and after treatment, figureFileSmall=tOTXH1PrmPUdK9XR4cMg6w==, figureFileBig=UFC3xfBRZb4ttRKEtJqoZQ==, tableContent=null), ArticleFig(id=1198589371738849771, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=图2, caption=1例IgG4相关性HIPT合并肝组织感染患者治疗前后肺CT对比

HIPT. 肝脏炎性假瘤

, figureFileSmall=tOTXH1PrmPUdK9XR4cMg6w==, figureFileBig=UFC3xfBRZb4ttRKEtJqoZQ==, tableContent=null), ArticleFig(id=1198589372858728946, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Fig.3, caption=Comparative abdominal contrast-enhanced MRI of nodules in liver segments S5 and S7 of 1 patient with IgG4-related HIPT and hepatic tissue infection before and one month after treatment, figureFileSmall=mFAhMJlkUZAJCEZzVq42Qw==, figureFileBig=gaOvWheowWFre8T+22FyBA==, tableContent=null), ArticleFig(id=1198589372984558073, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=图3, caption=1例IgG4相关性HIPT合并肝组织感染患者治疗前及治疗1个月后肝S5段、S7段结节腹部增强MRI对比

HIPT. 肝脏炎性假瘤

, figureFileSmall=mFAhMJlkUZAJCEZzVq42Qw==, figureFileBig=gaOvWheowWFre8T+22FyBA==, tableContent=null), ArticleFig(id=1198589373101998589, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Fig.4, caption=Hematoxylin and eosin (HE) staining and immunohistochemical staining of nodule in liver segment S5 in 1 patient with IgG4-related HIPT and hepatic tissue infection, figureFileSmall=x111t/Iui7nsS0q+LRi/6g==, figureFileBig=EUy8Vn4Xx5r0AnfOBzkiMQ==, tableContent=null), ArticleFig(id=1198589373223633411, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=图4, caption=1例IgG4相关性HIPT合并肝组织感染患者肝S5段结节HE及免疫组化染色

HIPT. 肝脏炎性假瘤；A. HE染色可见肝汇管区周围、肝组织内炎性细胞浸润(×100)；B. 免疫组化染色(×100)

, figureFileSmall=x111t/Iui7nsS0q+LRi/6g==, figureFileBig=EUy8Vn4Xx5r0AnfOBzkiMQ==, tableContent=null), ArticleFig(id=1198589373353656838, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Fig.5, caption=Comparison of abdominal contrast-enhanced MRI T1-weighted images of nodules in liver segments S5 and S7 of 1 patient with IgG4-related HIPT and hepatic tissue infection before and after treatment for 5 months, figureFileSmall=nD4lZ2moHWMItyTQ+aLs7Q==, figureFileBig=pR3+d5gqs83UYx1xjlc/kw==, tableContent=null), ArticleFig(id=1198589373454320136, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=图5, caption=1例IgG4相关性HIPT合并肝组织感染患者治疗前及治疗5个月后肝S5段、S7段结节腹部增强MRI T1序列比较

HIPT. 肝脏炎性假瘤

, figureFileSmall=nD4lZ2moHWMItyTQ+aLs7Q==, figureFileBig=pR3+d5gqs83UYx1xjlc/kw==, tableContent=null), ArticleFig(id=1198589373588537872, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=EN, label=Tab.1, caption=

Clinical imaging features of 26 cases of IgG4-related HIPT reported in the literature

, figureFileSmall=null, figureFileBig=null, tableContent=
研究

发表
时间

例数

年龄(岁)/

性别

血IgG4

(g/L)

CRP

(mg/L)

症状

HIPT

个数

大小位置活检

其余肿
大器官

影像转归
Zen等[3]2007667/男NANA肝功能异常单个3.4 cmNA有NANANA
Uchida等[4]2007154/男2.13NA黄疸，肝功能异常单个NA左叶有胰腺NA12个月复查消失
Naitoh等[5]2008177/男2.31NA腹痛，肝功能异常单个4.0 cm×3.0 cm左叶有无NA手术已切除
Ahn等[6]2011158/男NANANA单个3.0 cmNA有无NA手术已切除
Ahn等[6]2011160/男NANANA单个7.7 cmNA有无NA手术已切除
Ahn等[6]2011176/男NANANA两个2.3 cm/2.0 cmNA有无NA6个月复查缩小
Ahn等[6]2011152/男NANANA单个4.0 cmNA有胰腺NA43个月复查缩小
Kim等[7]2011158/男14.701腹痛单个NA左叶有肾边界清楚、均匀强化2个月复查缩小
Horiguchi等[8]2012176/男8.1914.3NA单个1.5 cmS2段有胆管边界清楚4个月复查缩小
Lee等[9]2013159/男0.75NA黄疸，肝功能异常单个NANA有胆管边界清楚、均匀强化1个月复查缩小
Matsuo等[10]2014174/男NA6无症状单个1.4 cmS8段有无边界清楚、均匀强化NA
Yang等[11]2015160/男15.90NA反酸单个NANA有食管、胃NANA
Mulki等[12]2015150/男NANA上腹痛两个6.6 cm/2.3 cm右叶有无边界清楚手术已切除
Shibata等[13]2016172/男1.3789.9无症状单个5.0 cmS7段有无边界清楚、环形强化6个月复查缩小
Miyajima等[14]2016150/女2.412恶心、胃痛单个NA后叶有无边界清楚、环形强化缩小
Legkiy等[15]2018160/男1.91NANA单个2.8 cmS2段有胰腺NABA
Jandee等[16]2020150/男NANA腹痛多个最大5.2 cm左、右叶有肺边界清楚、环形强化3个月复查缩小
Itazaki等[17]2021175/女5.24NA无症状单个2.5 cm左叶有胆道边界清楚、环形强化手术已切除
Hamano等[18]2021171/男1.80NA肝功能异常单个NAS3段有肾、胆道、淋巴结边界清楚1年复查缩小
Miyagi等[19]2022166/男NA4腹痛，肝功能异常单个3.5 cmS4段有无边界清楚、均匀强化1年后消失
Jang等[20]2022162/男7490.5背痛，肝功能异常单个6.0 cmS6段有胆道边界清楚6个月后消失
), ArticleFig(id=1198589373806641683, tenantId=1146029695717560320, journalId=1189873630562394117, articleId=1198558269032726836, language=CN, label=表1, caption=

文献报道的26例IgG4相关性HIPT患者的临床影像特征

, figureFileSmall=null, figureFileBig=null, tableContent=
研究

发表
时间

例数

年龄(岁)/

性别

血IgG4

(g/L)

CRP

(mg/L)

症状

HIPT

个数

大小位置活检

其余肿
大器官

影像转归
Zen等[3]2007667/男NANA肝功能异常单个3.4 cmNA有NANANA
Uchida等[4]2007154/男2.13NA黄疸，肝功能异常单个NA左叶有胰腺NA12个月复查消失
Naitoh等[5]2008177/男2.31NA腹痛，肝功能异常单个4.0 cm×3.0 cm左叶有无NA手术已切除
Ahn等[6]2011158/男NANANA单个3.0 cmNA有无NA手术已切除
Ahn等[6]2011160/男NANANA单个7.7 cmNA有无NA手术已切除
Ahn等[6]2011176/男NANANA两个2.3 cm/2.0 cmNA有无NA6个月复查缩小
Ahn等[6]2011152/男NANANA单个4.0 cmNA有胰腺NA43个月复查缩小
Kim等[7]2011158/男14.701腹痛单个NA左叶有肾边界清楚、均匀强化2个月复查缩小
Horiguchi等[8]2012176/男8.1914.3NA单个1.5 cmS2段有胆管边界清楚4个月复查缩小
Lee等[9]2013159/男0.75NA黄疸，肝功能异常单个NANA有胆管边界清楚、均匀强化1个月复查缩小
Matsuo等[10]2014174/男NA6无症状单个1.4 cmS8段有无边界清楚、均匀强化NA
Yang等[11]2015160/男15.90NA反酸单个NANA有食管、胃NANA
Mulki等[12]2015150/男NANA上腹痛两个6.6 cm/2.3 cm右叶有无边界清楚手术已切除
Shibata等[13]2016172/男1.3789.9无症状单个5.0 cmS7段有无边界清楚、环形强化6个月复查缩小
Miyajima等[14]2016150/女2.412恶心、胃痛单个NA后叶有无边界清楚、环形强化缩小
Legkiy等[15]2018160/男1.91NANA单个2.8 cmS2段有胰腺NABA
Jandee等[16]2020150/男NANA腹痛多个最大5.2 cm左、右叶有肺边界清楚、环形强化3个月复查缩小
Itazaki等[17]2021175/女5.24NA无症状单个2.5 cm左叶有胆道边界清楚、环形强化手术已切除
Hamano等[18]2021171/男1.80NA肝功能异常单个NAS3段有肾、胆道、淋巴结边界清楚1年复查缩小
Miyagi等[19]2022166/男NA4腹痛，肝功能异常单个3.5 cmS4段有无边界清楚、均匀强化1年后消失
Jang等[20]2022162/男7490.5背痛，肝功能异常单个6.0 cmS6段有胆道边界清楚6个月后消失
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IgG4相关性肝脏炎性假瘤合并肝组织感染1例并文献复习
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廖思敏 , 罗贵 , 金京玉 , 赵倩倩 , 朱剑 , 张江林 , 赵征 *
解放军医学杂志 | 临床研究 2024,49(7): 783-789
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解放军医学杂志 | 临床研究 2024, 49(7): 783-789
IgG4相关性肝脏炎性假瘤合并肝组织感染1例并文献复习
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廖思敏, 罗贵, 金京玉, 赵倩倩, 朱剑, 张江林, 赵征*
作者信息
  • 解放军总医院第一医学中心风湿免疫科，北京 100853

    • 廖思敏，医学博士，主治医师，主要从事结缔组织病及关节炎等疾病的诊治方面的研究

通讯作者:

赵征，E-mail：
IgG4-related hepatic inflammatory pseudotumor complicated with hepatic tissue infection: a case report and literature review
Si-Min Liao, Gui Luo, Jing-Yu Jin, Qian-Qian Zhao, Jian Zhu, Jiang-Lin Zhang, Zheng Zhao*
Affiliations
  • Department of Rheumatology and Immunology, the First Medical Center of Chinese PLA General Hospital, Beijing 100853, China
出版时间: 2024-07-28 doi: 10.11855/j.issn.0577-7402.1209.2024.0512
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目的 报道1例以多发肺、肝结节为临床表现的IgG4相关性肝脏炎性假瘤(HIPT)合并肝组织感染患者的临床影像特点并复习文献，以期帮助临床医师诊治及鉴别IgG4相关性肝脏炎性假瘤。方法 回顾性分析1例多发肺、肝结节的IgG4相关性疾病(IgG4-RD)患者的病例资料，并通过检索中英文数据库，综合文献结果，总结IgG4相关性HIPT和肝组织感染的临床影像特点。结果 本例为64岁女性，因“纳差乏力1年余、干咳4个月”至解放军总医院第一医学中心风湿免疫科住院，仅有肺、肝多发结节，无眼睑、腮腺、颌下腺、胰腺等受累，血IgG4 (14.1 g/L)及C反应蛋白(CRP，82.1 mg/L)水平均升高。肺CT提示双肺多发实性结节、边界清楚。腹部增强磁共振检查显示肝S7段结节，周围有假包膜，边界清楚，均匀强化；肝S5段结节，边界模糊，环形强化；最终肝结节通过病理和病原学宏基因测序证实S7段结节为IgG4相关性HIPT，S5段结节为肝组织感染。经足疗程抗感染和甲泼尼龙、来氟米特治疗后，复查肺、肝结节基本消失。检索中国知网、万方数据知识服务平台、PubMed数据库(截至2023年9月)，未见IgG4-RD肝脏受累同时合并感染的病例报道。目前共报道26例IgG4-RD肝脏受累病例，男性居多(92.3%)，平均年龄51岁，多以肝功能异常为首发症状就诊，血炎症指标正常，影像表现以单个结节多见(88.5%)，边界清楚，均匀强化和环形强化均可见，常同时合并胰腺、胆道受累，病理为确诊该疾病的金标准。结论 本例肝内多发结节为IgG4相关性HIPT与感染共存。在诊治以肝多发结节为表现的IgG4-RD患者时，若结节影像学特征不一致，需考虑原发病与其他情况并存的情况，该病容易误诊，须获取病理组织帮助确诊。

IgG4相关性疾病  /  肝脏炎性假瘤  /  感染

Objective To report the clinical and imaging characteristics of a case with the clinical manifestation of multiple lung and liver nodules, diagnosed as IgG4-related hepatic inflammatory pseudotumor (HIPT) complicated by hepatic tissue-infection, and review the literature in order to facilitate clinical diagnosis and differential identification of IgG4-related HIPT. Methods A retrospective analysis was conducted on the medical records of a patient with IgG4-related disease (IgG4-RD) characterized by multiple lung and liver nodules. A literature review was performed by searching Chinese and English databases to summarize the clinical and imaging characteristics of IgG4-related HIPT and hepatic tissue infection. Results The case involved a 64-year-old female admitted to the Rheumatology and Immunology Department of the First Medical Center of Chinese PLA General Hospital due to "poor appetite and fatigue for over a year, and dry cough for four months". She presented with multiple nodules in the lungs and liver, without involvement of the eyelids, salivary glands, submandibular glands, or pancreas. Laboratory test results revealed elevated serum IgG4 levels at 14.1 g/L and C-reactive protein (CRP) at 82.1 mg/L. Pulmonary CT scans indicated multiple solid nodules in both lungs with clear boundaries. Abdominal contrast-enhanced MRI revealed a nodule in liver segment S7 with a pseudocapsule around it, clear boundaries, and uniform enhancement; another nodule in liver segment S5 with blurred boundaries and ring enhancement. The final diagnosis of the liver nodules was confirmed by pathological and metagenomic sequencing to be an IgG4-related HIPT in segment S7 and hepatic tissue infection in segment S5. After a full course of anti-infection and treatment with methylprednisolone and leflunomide, follow-up imaging showed near-complete resolution of the lung and liver nodules. Literatures were searched in China National Knowledge Infrastructure (CNKI), Wanfang, and PubMed databases (up to September 2023), and no case of IgG4-RD complicated with both liver involvement and infection was found. A total of 26 cases of IgG4-RD involving the liver have been reported so far, predominantly in males (92.3%), with an average age of 51 years. Most patients presented with abnormal liver function as the initial symptom, with normal blood inflammatory markers. Imaging typically shows a single nodule in 88.5% of cases, with clear boundaries and uniform enhancement, as well as ring enhancement. Concurrent involvement of the pancreas and biliary tract is common. Pathology is the gold standard for confirming the disease. Conclusions This case reports coexistence of IgG4-related HIPT and infection within multiple hepatic nodules. In the diagnosis and treatment of patients with IgG4-RD presenting with multiple hepatic nodules, if the imaging characteristics of the nodules are inconsistent, it is necessary to consider the possibility of the underlying disease coexisting with other conditions, which can be easily misdiagnosed. Actively obtaining pathological tissue is crucial for aiding in the definitive diagnosis.

IgG4-related disease  /  hepatic inflammatory pseudotumor  /  infection
廖思敏, 罗贵, 金京玉, 赵倩倩, 朱剑, 张江林, 赵征. IgG4相关性肝脏炎性假瘤合并肝组织感染1例并文献复习. 解放军医学杂志, 2024 , 49 (7) : 783 -789 . DOI: 10.11855/j.issn.0577-7402.1209.2024.0512
Si-Min Liao, Gui Luo, Jing-Yu Jin, Qian-Qian Zhao, Jian Zhu, Jiang-Lin Zhang, Zheng Zhao. IgG4-related hepatic inflammatory pseudotumor complicated with hepatic tissue infection: a case report and literature review[J]. Medical Journal of Chinese People’s Liberation Army, 2024 , 49 (7) : 783 -789 . DOI: 10.11855/j.issn.0577-7402.1209.2024.0512
正文
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IgG4相关性疾病(IgG4-RD)是一类以IgG4阳性浆细胞浸润组织导致器官肿大的慢性自身免疫性疾病，胰腺、胆道、涎腺、腹膜后等为常见受累部位[1]。肝脏受累少见，可表现为自身免疫性肝病和肝脏炎性假瘤(hepatic inflammatory pseudotumor，HIPT)两种形式[2]。本文报道1例IgG4-RD肺、肝多发结节，肝结节经病理及病原学宏基因测序显示为HIPT合并感染的病例，并总结既往国内外详细报道的IgG4相关性HIPT患者的临床影像特点，以期提高临床医师对IgG4-RD合并肝多发结节的诊治鉴别思路和认识。
1 病例资料
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1.1 病史
患者女，64岁，因“食欲缺乏、乏力1年余，干咳4个月”于2022年9月收入解放军总医院第一医学中心风湿免疫科。患者2021年5月无明显诱因出现食欲缺乏、乏力，无恶心、呕吐、腹痛，当地医院实验室检查白细胞10.99×109/L、红细胞4.28×109/L、血小板357×109/L、红细胞沉降率93 mm/h、免疫球蛋白IgG 33 g/L、C反应蛋白(CRP)44.6 mg/L。肺CT检查提示两肺上叶及右肺中叶小结节灶，右肺下叶及中叶炎症，肝脏低密度灶。当地医院诊断“免疫球蛋白升高；肺结节”，给予患者祛痰、镇咳、护胃等对症治疗，食欲缺乏、乏力未见明显缓解。2022年5月无明显诱因出现干咳，晨起明显，仍有明显食欲缺乏、乏力，无发热。当地医院检查CRP 56.8 mg/L、甲胎蛋白正常。给予患者“青霉素”治疗后，食欲缺乏、乏力稍有缓解。2022年7月转诊至北京某三甲医院，查白细胞8.3×109/L、CRP 42.8 mg/L；上腹部磁共振成像(MRI)显示肝右叶肿块周围多发异常信号，考虑：(1)原发性肝癌？(2)感染性病变？正电子发射计算机断层显像(PET-CT)显示肝右后叶肿块，氟代脱氧葡萄糖(FDG)代谢明显增高；两肺多发间质性病变(胸膜下多发结节、中央间质增厚、小叶间隔增厚)，考虑：(1)肝脏及两肺病变均为淋巴瘤；(2)肝脏及两肺病变均为结节病；(3)不除外肝脏其他恶性肿瘤并两肺转移(淋巴道播散)。2022年7月完善超声引导下肝穿刺活检术(S7段)，术后病理提示肝组织内见大量浆细胞、中性粒细胞、淋巴细胞浸润伴间质纤维组织增生。免疫组化：甲胎蛋白(AFP)(-)、CD34(脉管+)、CD38+、CD138+、Ki-67(10%)、P53(野生型)、κ+、λ+、IgG4+、IgG+、IgG4+[>30个/高倍镜视野(HPF)%]、IgG4/IgG+细胞>40%(图1)。
该例患者于2022年9月转诊至本科门诊，查IgG 60.8 g/L、IgG1 39.7 g/L、IgG2 9.7 g/L、IgG3 1.5 g/L、IgG4 14.1 g/L，门诊以“IgG4-RD可能性大”收入院，并给予甲泼尼龙8 mg/d、来氟米特10 mg/d、硫酸羟氯喹0.2 g×2次/d治疗。患者病程中无眼睑、腮腺肿大，无腹痛，无关节肿痛、皮疹等不适。无特殊既往史、个人史、家族史。入院体检：体温36.5 ℃、脉搏78次/min、呼吸20次/min、血压132/68 mmHg，全身未触及浅表淋巴结肿大，心肺腹、神经系统查体未见异常。
1.2 辅助检查
入院后查IgG1 30.4 g/L、IgG2 8.9 g/L、IgG3 0.9 g/L、IgG4 11.5 g/L，补体C3 0.8 g/L、C4 0.2 g/L、IgG 51.6 g/L，白细胞8.61×109/L、红细胞3.91×109/L、血小板276×109/L，CRP 82.1 mg/L，肝肾功能、电解质、抗核抗体谱、自身抗体谱、肿瘤标志物、真菌D-葡聚糖(G试验)、曲霉菌半乳甘露聚糖检测(GM试验)、呼吸道病原学、巨细胞病毒、EB病毒、结核杆菌感染T细胞斑点试验(T-SPOT)、血和尿免疫电泳、痰涂片、痰培养、涎腺超声均未见异常。
1.3 影像学检查
患者治疗前，肺部CT显示：双肺多发结节，不除外转移瘤的可能；双肺间质性改变伴多发炎症(图2)。腹部增强MRI见图3，结果显示：(1)S7段肝右叶上端见长T2长T1信号类圆形肿块，边界清楚，磁共振弥散加权成像(DWI)呈明显高信号，相应区域ADC信号明显减低，反相位图像病变未见降低。动态增强扫描病变动脉期明显均匀强化，门脉期及延迟期轮廓清晰，可见假包膜。(2)S5段肝右叶前下段近包膜可见长T2长T1信号，边缘模糊呈稍长T2稍长T1信号，病变DWI呈明显高信号，ADC信号减低，反相位病变未见信号减低，增强扫描动脉期明显环形强化，门脉期及延迟期持续边缘轻度强化，中心未见强化。考虑：(1)肝右后叶上段肿块，考虑非肿瘤性病变，IgG4-RD可能性大；(2)肝右叶前下段近包膜下异常信号，考虑感染性病变。
1.4 诊断、治疗及随访
鉴于患者肝内两段均存在结节，且影像增强方式不同，外院已行S7段穿刺活检，故予以行S5段穿刺及组织病原学宏基因测序(NGS)，以明确结节性质。S5段HE染色结果显示，肝细胞浊肿，汇管区慢性炎性细胞浸润，局灶急慢性炎性细胞浸润(图4A)；免疫组化结果显示偶见CD138+、CD38+、IgG4+细胞、IgG(部分细胞+)，IgG4+/IgG+细胞<40%(图4B)。肝组织(S5段)NGS结果显示牙龈卟啉单胞菌(序列数2584)、光滑念珠菌(序列数380)。
根据入院后完善的实验室检查，诊断不考虑肿瘤性疾病。患者肺结节及肝S7段结节诊断IgG4-RD；肝S5段结节诊断肝组织感染(牙龈卟啉单胞菌、光滑念珠菌)。治疗调整为甲泼尼龙40 mg/d、来氟米特10 mg/d、硫酸羟氯喹0.2 g 2次/d，并加用哌拉西林他唑巴坦、氟康唑、甲硝唑抗感染治疗。1周后复查血IgG4 6.7 g/L，CRP 3.8 mg/L。治疗1个月后复查肺CT，可见结节明显缩小，部分结节消失(图2)；复查腹部增强MRI可见S5、S7段结节均有缩小(图3)。出院后患者继续口服氟康唑，5个月后复查腹部增强MRI可见S5段病变基本消失，S7段结节明显缩小(图5)。
2 文献检索及复习
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在中国知网、万方数据知识服务平台中，以中文“IgG4相关性疾病”“炎性假瘤”“肝”为关键词未检索出中文文献；以中文“IgG4相关性疾病”“肺结节”“肝结节”为关键词未检索出中文文献；以中文“IgG4相关性疾病”“炎性假瘤”“感染”为关键词未检索出中文文献。在PubMed数据库中，以“IgG4-related disease”“inflammatory pseudotumor”“liver”为关键词共检索出20篇文献，其中2篇为日文，摘要及全文无法获取；另外18篇为英文，共26例病理证实的IgG4相关性肝脏炎性假瘤患者[3-20]。以“IgG4-related disease”“inflammatory pseudotumor”“infection”为关键词未检索出英文文献。
表1总结了文献报道的26例IgG4相关性HIPT患者的临床影像特征。其中男24例(92.3%)，女2例(7.7%)；平均年龄51岁(50~76岁)；12例检测血IgG4水平的患者中，11例(91.7%)血IgG4水平升高；仅1例血CRP水平升高，其余患者CRP均正常。46.7%的患者出现肝功能异常；其余临床表现有腹痛、背痛、黄疸、反酸、恶心；3例患者无症状。影像学检查发现，IgG4相关性HIPT大部分为肝内单发结节(88.5%)；左、右叶均有报道；边界清楚；增强MRI检查显示有50%均匀强化、50%有环形强化。26例IgG4相关性HIPT患者中，仅肝脏受累9例(34.6%)；合并其他器官肿大11例，其中胰腺受累3例，肾脏受累1例，胆管受累4例，肺受累1例，合并食管和胃受累1例，合并肾、胆道和淋巴结受累1例。
3 讨论
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IgG4-RD是由免疫介导的组织慢性炎症伴纤维化的一类隐匿进展性自身免疫性疾病，临床表现以器官或组织肿大为特征，常见受累器官或部位为泪腺、唾液腺、胰腺、胆管、腹膜后，而肺、肝脏受累较少[1]。IgG4相关肺疾病(IgG4-RLD)于2004年首次被报道[21]，患者可出现咳嗽、咳痰、咯血、胸痛、呼吸困难等症状，易被误诊为肺部肿瘤性疾病[22]。IgG4-RLD多以个案报道居多，大样本量的横断面研究极少[23]。日本学者提出将累及胸腔内器官及辅助结构组织的病变统称为IgG4-RLD，包括肺实质、肺间质、气道、纵隔、胸膜和肺血管[24]。IgG4-RLD一般多与肺外表现同时出现，少数情况以首发症状单独累及肺。张盼盼等[25]对346例IgG4-RD患者器官受累情况分析发现，最常见的受累器官为颌下腺，仅28%为肺受累。IgG4-RLD胸部CT表现分为4种类型：(1)实性结节型，单发结节，包括肿块；(2)圆形磨玻璃型；(3)肺泡间质型，表现为蜂窝样改变、支气管扩张、弥漫磨玻璃影；(4)支气管血管束型，表现为支气管血管束增厚和小叶间隔增厚，部分患者可伴有肺门淋巴结肿大[26]。Xie等[27]报道明确诊断为IgG4-RD中50例合并肺结节患者的肺部影像学特点，发现48例(96%)肺结节直径<1.2 cm，其中多发39例(78%)，两肺多见34例(68%)，肺野内随机分布32例(64%)；绝大部分肺结节为实性结节46例(92%)，边界清楚44例(88%)，32例(64%)可伴纵隔和肺门淋巴结肿大，31例(62%)对治疗有反应。本例患者肺结节呈多发、两肺分布、边界清楚的实性小结节，均与报道的IgG4-RLD中肺结节性质相符；虽然未能取得肺结节病理，但激素和免疫抑制剂治疗后部分结节消失，考虑该患者肺部多发结节与IgG4-RD相关。
本例另一表现为肝结节，分别位于肝脏S5段和S7段，二者性质不同。尽管S7段结节病理明确提示无肿瘤细胞，有IgG4-RD典型病理表现，但S7段结节边界规则，有假包膜，增强MRI显示均匀强化，S5段结节边界模糊，增强MRI显示环形强化，两处结节在影像学的表现不完全一致，增加了诊断的迷惑性。IgG4-RD肝脏受累各国队列报道比例不一，但占比均非常低。Inoue等[28]回顾分析了多中心共235例IgG4-RD患者的临床特征，未发现有肝脏受累的病例；张盼盼等[25]报道的346例IgG4-RD病例中亦无肝脏受累患者；而Wallace等[29]报道的125例病理确诊为IgG4-RD患者中，肝脏受累仅3例(2.4%)。IgG4-RD肝脏受累相对罕见，目前认为可分为两种类型：(1)自身免疫性肝病，临床可表现为谷丙转氨酶、谷草转氨酶、γ-谷氨酰转移酶、胆红素等异常升高、血清IgG4水平升高，病理表现为小叶性肝炎、门脉炎症、胆管损伤等；(2)HIPT，定义为肝实质或肝门的结节性病变，主要位于肝右叶，边界清楚，病理表现为大量IgG4阳性浆细胞浸润伴闭塞性脉管炎[2]。结合病理和影像，本例患者位于肝脏S7段的病变属于IgG4相关性HIPT。IgG4相关性HIPT十分罕见，查阅国内外文献，目前共有26例个案报道，绝大部分为男性，而本研究报道了1例64岁女性病例。本病例两处肝结节影像表现不同，诊断时存在难点，且其中S5段结节边界模糊，与典型的IgG4相关性HIPT表现不同；加之患者的血CRP水平多次升高，故需鉴别其他疾病的可能。张霞等[30]提出，当临床上疑诊IgG4-RD的患者出现反复发热、糖皮质激素治疗无改善、CRP水平升高5倍以上、影像学提示有坏死、空洞、分隔性腹部或盆腔积液均需与感染重点鉴别。本病例多次查血CRP水平升高且在5倍以上，虽然无腹痛、发热等症状，但完善S5段肝结节穿刺送检NGS，结果显示细菌合并真菌感染。本例在应用糖皮质激素、来氟米特后，复查血IgG4水平下降，但CRP水平仍继续升高；在抗细菌、真菌治疗后，CRP水平恢复正常。5个月足疗程抗感染后，复查MRI可见S5段的感染性病变基本消失，S7段的IPT明显缩小，提示治疗有效。
综上所述，IgG4-RD可出现肺结节和肝结节，肺结节以多发、双侧、边界清楚的实性小结节多见，肝结节则以单个、边界清楚的HIPT多见，二者对激素和免疫抑制剂效果均较好。本病例在肝脏同时存在IgG4-RD和感染性结节性病变，比较少见。临床上对于多发肝脏结节性病变，单纯从影像学增强方式判断病变性质容易误诊，尤其是存在多处结节，且结节的影像学特征不完全一致时，需尽量获取病理组织学标本，以帮助诊断IgG4相关性HIPT，同时可鉴别与IgG4-RD有相似临床或影像表现的其他疾病。
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2024年第49卷第7期
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doi: 10.11855/j.issn.0577-7402.1209.2024.0512
  • 接收时间：2023-09-12
  • 首发时间：2025-11-21
  • 出版时间：2024-07-28
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  • 收稿日期：2023-09-12
  • 录用日期：2023-10-16
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    解放军总医院第一医学中心风湿免疫科，北京 100853

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鹅膏菌科Amanitaceae 2 11 5.26 鹅膏菌属 Amanita 10 4.78
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