Pregnancy complicated with IgG4-related thyroid disease: a case report and literature review

doi:10.3877/cma.j.issn.2095-3259.2024.01.007

Abstract

Abstract:

Objective

This study is a case report of a patient with immunoglobulin-G₄ related disease (IgG₄-RD) associated with pregnancy. It aims to provide a comprehensive analysis of the patient′s clinical data, diagnosis, treatment, and follow-up outcomes, to enhance awareness among obstetricians regarding the significance of IgG₄-RD.

Methods

A retrospective analysis was conducted on a patient with pregnancy complicated by IgG₄-RD, who was admitted to Peking University Shenzhen Hospital on January 18, 2023. A thorough literature search was performed on pregnancy-related IgG₄-RD case reports in the web databases.

Results

A 29-year-old patient with hyperthyroidism for 4⁺ years and IgG₄-RD for 7 months was admitted to our hospital with twin pregnancy at 29 weeks of gestation due to hyperthyroidism crisis, grade IV cardiac function, IgG₄-related disease, preeclampsia, and novel coronavirus pneumonia virus infection. A multidisciplinary consultation treatment was taken on her and achieved good perinatal outcomes. A total of six (including current case) case reports regarding pregnancy with IgG₄-RD were identified, which exhibited varying symptoms, affected organs, and clinical presentations. All cases showed symptom improvement after receiving standardized hormone treatment.

Conclusions

IgG₄-RD related manifestations during pregnancy are intricate, potentially impacting vital organ function. Women with IgG₄-related thyroid disease can achieve successful pregnancy, but there is a high risk of pregnancy. Continuous hormone maintenance therapy and multidisciplinary close monitoring are needed during and after pregnancy.

Key words: Immunoglobulin G₄-related disease, Hyperthyroidism, Pregnancy, Postpartum period

Qianwen Cui, Yanlan Wang, Qing Li, Pingyue Zhao, Yifei Xu, Zhidong Yuan, Wei Han, Xiaowei Zhang, Lei Huang, Shangrong Fan. Pregnancy complicated with IgG₄-related thyroid disease: a case report and literature review[J]. Chinese Journal of Obstetric Emergency(Electronic Edition), 2024, 13(01): 33-40.

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Figures/Tables 6

References 27

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病例	年龄(岁)	发病时间	主要症状	主要诊断	诊断时间	血清IgG₄	重要实验室检查
A	23	孕33周	上腹痛、放射至背部，恶心，呕吐	IgG₄相关胆管病，自身免疫性胰腺炎	产后	165.0 mg/dl↑	血清淀粉酶106 U/L↑，天冬氨酸转氨酶66 U/L↑，丙氨酸转氨酶80 U/L↑，碱性磷酸酶730 U/L↑
B	25	产后21 d	头痛，乏力，视觉障碍	IgG₄相关垂体炎	产后	55.7 mg/dl	硫酸脱氢表雄酮3 μg/dl↓，促肾上腺皮质激素<5.0 pg/ml↓，皮质醇<2.0 μg/dl↓，垂体及靶激素水平较低
C	24	产后21 d	干咳，发热，呼吸困难	IgG₄相关孤立性肺疾病	产后	－	血沉105 mm/h↑，C反应蛋白28.74 mg/L↑
D	40	孕32周	发热，胸痛，呼吸困难，视觉障碍，头痛	IgG₄相关淋巴细胞性腺垂体炎伴Rathke裂囊肿	妊娠期	－	促肾上腺皮质激素<5 pg/ml↓，黄体生成素<0.5 mlU/ml↓，卵泡刺激素<0.5 mIU/ml↓，催乳素<1.0 ng/ml↓，皮质醇<1.0 μg/dl↓
E	25	产后120 d	眼睑肿胀、发红，球后疼痛，面部结节	IgG₄相关眼病	产后	－	甲状腺功能相关指标正常
本例	29	孕29^＋1周	下肢水肿，呼吸困难，心悸	IgG₄-RTD^[10]，甲状腺危象	妊娠期	3889.4 mg/dl↑	促甲状腺激素>0.005 mIU/L↓，FT₄-国际单位18.85 pmol/L↑，甲状腺过氧化物酶抗体665.6 IU/ml↑，甲状腺球蛋白抗体871.9 IU/ml↑，甲状腺微粒体抗体>1000.0 IU/ml↑，促甲状腺受体抗体>30 IU/L↑

病例	年龄(岁)	发病时间	主要症状	主要诊断	诊断时间	血清IgG₄	重要实验室检查
A	23	孕33周	上腹痛、放射至背部，恶心，呕吐	IgG₄相关胆管病，自身免疫性胰腺炎	产后	165.0 mg/dl↑	血清淀粉酶106 U/L↑，天冬氨酸转氨酶66 U/L↑，丙氨酸转氨酶80 U/L↑，碱性磷酸酶730 U/L↑
B	25	产后21 d	头痛，乏力，视觉障碍	IgG₄相关垂体炎	产后	55.7 mg/dl	硫酸脱氢表雄酮3 μg/dl↓，促肾上腺皮质激素<5.0 pg/ml↓，皮质醇<2.0 μg/dl↓，垂体及靶激素水平较低
C	24	产后21 d	干咳，发热，呼吸困难	IgG₄相关孤立性肺疾病	产后	－	血沉105 mm/h↑，C反应蛋白28.74 mg/L↑
D	40	孕32周	发热，胸痛，呼吸困难，视觉障碍，头痛	IgG₄相关淋巴细胞性腺垂体炎伴Rathke裂囊肿	妊娠期	－	促肾上腺皮质激素<5 pg/ml↓，黄体生成素<0.5 mlU/ml↓，卵泡刺激素<0.5 mIU/ml↓，催乳素<1.0 ng/ml↓，皮质醇<1.0 μg/dl↓
E	25	产后120 d	眼睑肿胀、发红，球后疼痛，面部结节	IgG₄相关眼病	产后	－	甲状腺功能相关指标正常
本例	29	孕29^＋1周	下肢水肿，呼吸困难，心悸	IgG₄-RTD^[10]，甲状腺危象	妊娠期	3889.4 mg/dl↑	促甲状腺激素>0.005 mIU/L↓，FT₄-国际单位18.85 pmol/L↑，甲状腺过氧化物酶抗体665.6 IU/ml↑，甲状腺球蛋白抗体871.9 IU/ml↑，甲状腺微粒体抗体>1000.0 IU/ml↑，促甲状腺受体抗体>30 IU/L↑

病例	影像学检查	病理组织学
A	腹部超声、MRCP提示胰管远端扩张，胰头增大、疑似病变；胆总管扩张	－
B	增强MRI提示垂体、垂体柄弥漫性增强、扩大	经蝶垂体活检病理：大量淋巴细胞浸润、席纹状纤维化，见219个/每高倍镜视野IgG^＋细胞；免疫组化IgG₄^＋浆细胞48个/每高倍视野
C	胸部X线和CT提示双肺多发肺实变	TBLB病理：肺泡间隔纤维化，浆细胞及淋巴细胞浸润；VATS肺活检：密集淋巴浆细胞浸润，呈层状纤维化；IgG₄^＋/IgG^＋为60%
D	CT提示实性垂体肿块均匀增强，鞍上延伸；两月后CT图像变为部分囊性肿瘤增大；MRI提示鞍内和鞍上部分囊性病变压迫视交叉	经蝶入路部分病灶切除术病理：淋巴细胞弥漫性广泛浸润，纤维组织增生；免疫组化见IgG₄^＋浆细胞簇
E	眼眶MRI提示外侧直肌和上直肌增大，眼睑水肿，泪腺肿胀	右泪腺活检病理：CD138和IgG₄免疫组化染色显示浆细胞浸润
本例	超声：甲亢声像；唾液腺回声减低；胸片：心影增大，左侧胸腔少量积液可能；PET-CT：垂体窝不均匀代谢活性增高灶；甲状腺体积明显增大伴弥漫性代谢活性稍高；甲亢性心脏病；右侧胸腔积液	－

病例	影像学检查	病理组织学
A	腹部超声、MRCP提示胰管远端扩张，胰头增大、疑似病变；胆总管扩张	－
B	增强MRI提示垂体、垂体柄弥漫性增强、扩大	经蝶垂体活检病理：大量淋巴细胞浸润、席纹状纤维化，见219个/每高倍镜视野IgG^＋细胞；免疫组化IgG₄^＋浆细胞48个/每高倍视野
C	胸部X线和CT提示双肺多发肺实变	TBLB病理：肺泡间隔纤维化，浆细胞及淋巴细胞浸润；VATS肺活检：密集淋巴浆细胞浸润，呈层状纤维化；IgG₄^＋/IgG^＋为60%
D	CT提示实性垂体肿块均匀增强，鞍上延伸；两月后CT图像变为部分囊性肿瘤增大；MRI提示鞍内和鞍上部分囊性病变压迫视交叉	经蝶入路部分病灶切除术病理：淋巴细胞弥漫性广泛浸润，纤维组织增生；免疫组化见IgG₄^＋浆细胞簇
E	眼眶MRI提示外侧直肌和上直肌增大，眼睑水肿，泪腺肿胀	右泪腺活检病理：CD138和IgG₄免疫组化染色显示浆细胞浸润
本例	超声：甲亢声像；唾液腺回声减低；胸片：心影增大，左侧胸腔少量积液可能；PET-CT：垂体窝不均匀代谢活性增高灶；甲状腺体积明显增大伴弥漫性代谢活性稍高；甲亢性心脏病；右侧胸腔积液	－

病例	主要治疗	围产结局	随访
A	泼尼松40 mg/d，6个月内逐渐减少剂量	38^＋4周阴道分娩，新生儿健康	患者自行停药导致复发
B	泼尼松40 mg/d，逐渐减少剂量至月经复潮，改用小剂量(10 mg/d)氢化可的松	产后发病	无复发
C	皮质类固醇60 mg/d，1个月后减至50 mg/d，后每月减少10 mg/d，直至停药	产后发病	无复发
D	经蝶入路部分病灶切除术；氢化可的松激素替代	足月阴道分娩，新生儿健康	－
E	甲泼尼龙16 mg/d，后逐渐减少剂量	产后发病	激素逐渐减量过程中症状复发；予第2疗程甲泼尼龙；继而随访2年内无症状复发，有IgG₄水平上升；随访第3年出现周期性皮疹
本例	孕期：泼尼松10-5-10 mg 3次/d；产前：地塞米松20 mg(12 h重复1次，共2次)；择期剖宫产终止妊娠；产后：泼尼松10 mg 3次/d	剖宫产分娩早产儿，健康	继续抗甲亢治疗，激素逐渐减量维持治疗。监测血清IgG₄较前降低，最低809.2 mg/L，甲状腺功能指标在正常范围

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