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孕期大疱性皮肤病变的一种不寻常表现。

An Unusual Presentation of Bullous Skin Lesions During Pregnancy.

作者信息

Jordan Stanley C, Balzer Bonnie, Morrison Susane, Truong Allison, Karumanchi Ananth

机构信息

Department of Medicine, Cedars-Sinai Medical Center, Los Angeles, CA, USA.

Department of Pathology, Cedars-Sinai Medical Center, Los Angeles, CA, USA.

出版信息

J Investig Med High Impact Case Rep. 2025 Jan-Dec;13:23247096251347403. doi: 10.1177/23247096251347403. Epub 2025 Jun 12.

DOI:10.1177/23247096251347403
PMID:40503690
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12163261/
Abstract

Here, we present a 33-years-old pregnant female who at 27-weeks gestation developed an unusual rash on her left knee after a spider bite. The rash rapidly became systemic requiring admission and treatment with high-dose steroids. Patient rapidly developed complications of steroid therapy and saw no benefits in reducing the activity of the rash of symptoms of itching. A repeat biopsy showed linear fluorescence along the dermal epidermal junction with C3 (strong) and IgG (weak) suggestive of pemphigus gestationis (PG). After balancing concerns for the health of the mother and risk for disease transmission to the child, we decided to treat the patient with high-dose intravenous immunoglobulin (IVIg). This resulted in rapid remission of disease and delivery of a healthy child without evidence of PG at 36-weeks gestation. A discussion of the mechanism(s) of action of IVIg that were salutary in this case is presented. Importantly, we discuss the likely benefits of IVIg in saturation of the Fc-receptor neonatal IgG recycling and preservation system in accelerating the degradation of pathogenic IgGs and the inhibitory of effects of IVIg on C3 activation which was the predominant immunoreactant in skin biopsy. The patient recovered completely after the fourth monthly IVIg infusion and continues to do well.

摘要

在此,我们报告一位33岁的孕妇,她在妊娠27周时被蜘蛛叮咬后左膝出现了一种不寻常的皮疹。皮疹迅速蔓延至全身,需要住院并接受大剂量类固醇治疗。患者迅速出现了类固醇治疗的并发症,且在减轻皮疹活动或瘙痒症状方面未见益处。再次活检显示沿真皮表皮交界处有线性荧光,C3(强)和IgG(弱)阳性,提示妊娠类天疱疮(PG)。在权衡对母亲健康的担忧和疾病传染给胎儿的风险后,我们决定用大剂量静脉注射免疫球蛋白(IVIg)治疗该患者。这导致疾病迅速缓解,并在妊娠36周时顺利产下一名健康婴儿,且未发现PG迹象。本文讨论了在该病例中有益的IVIg作用机制。重要的是,我们讨论了IVIg在饱和Fc受体新生儿IgG再循环和保存系统方面的可能益处,这有助于加速致病性IgG的降解,以及IVIg对C3激活的抑制作用,而C3激活是皮肤活检中的主要免疫反应物。患者在第四次每月一次的IVIg输注后完全康复,目前情况良好。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/cf3ecfccd972/10.1177_23247096251347403-fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/cf29b306fc2f/10.1177_23247096251347403-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/009699b41dba/10.1177_23247096251347403-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/cf3ecfccd972/10.1177_23247096251347403-fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/cf29b306fc2f/10.1177_23247096251347403-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/009699b41dba/10.1177_23247096251347403-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f2f0/12163261/cf3ecfccd972/10.1177_23247096251347403-fig3.jpg

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1
An Unusual Presentation of Bullous Skin Lesions During Pregnancy.孕期大疱性皮肤病变的一种不寻常表现。
J Investig Med High Impact Case Rep. 2025 Jan-Dec;13:23247096251347403. doi: 10.1177/23247096251347403. Epub 2025 Jun 12.
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本文引用的文献

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Efgartigimod: A Review in Generalised Myasthenia Gravis.依氟鸟氨酸:一种全身性重症肌无力的综述。
Drugs. 2024 Nov;84(11):1463-1474. doi: 10.1007/s40265-024-02101-9. Epub 2024 Nov 7.
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Binding to the neonatal Fc receptor enhances the pathogenicity of anti-desmoglein-3 antibodies in keratinocytes.与新生儿 Fc 受体结合可增强角质形成细胞中抗桥粒芯糖蛋白-3 抗体的致病性。
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State-of-the-art diagnosis of autoimmune blistering diseases.自身免疫性水疱病的最新诊断方法。
Front Immunol. 2024 Jun 6;15:1363032. doi: 10.3389/fimmu.2024.1363032. eCollection 2024.
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Diagnosis, fetal risk and treatment of pemphigoid gestationis in pregnancy: A case report.妊娠期类天疱疮的诊断、胎儿风险及治疗:一例病例报告。
World J Clin Cases. 2021 Dec 6;9(34):10645-10651. doi: 10.12998/wjcc.v9.i34.10645.
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Intravenous immunoglobulin significantly reduces exposure of concomitantly administered anti-C5 monoclonal antibody tesidolumab.静脉注射免疫球蛋白可显著降低同时给药的抗C5单克隆抗体替西多单抗的暴露量。
Am J Transplant. 2020 Sep;20(9):2581-2588. doi: 10.1111/ajt.15922. Epub 2020 May 13.
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Autoimmune bullous skin diseases, pemphigus and pemphigoid.自身免疫性大疱性皮肤病,天疱疮和类天疱疮。
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Implications of Fc Neonatal Receptor (FcRn) Manipulations for Transplant Immunotherapeutics.Fc 新生儿受体(FcRn)调控对移植免疫治疗的影响。
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Autoimmune bullous diseases during pregnancy: insight into pathogenetic mechanisms and clinical features.自身免疫性大疱性疾病在妊娠期间的表现:对发病机制和临床特征的深入了解。
G Ital Dermatol Venereol. 2019 Jun;154(3):256-262. doi: 10.23736/S0392-0488.18.06153-9. Epub 2018 Oct 29.
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Pemphigoid gestationis successfully treated with intravenous immunoglobulin.静脉注射免疫球蛋白成功治疗妊娠类天疱疮。
BMJ Case Rep. 2018 Apr 7;2018:bcr-2018-224346. doi: 10.1136/bcr-2018-224346.
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Update on the use of immunoglobulin in human disease: A review of evidence.免疫球蛋白在人类疾病中的应用更新:证据回顾。
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