Jordan Stanley C, Balzer Bonnie, Morrison Susane, Truong Allison, Karumanchi Ananth
Department of Medicine, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
Department of Pathology, Cedars-Sinai Medical Center, Los Angeles, CA, USA.
J Investig Med High Impact Case Rep. 2025 Jan-Dec;13:23247096251347403. doi: 10.1177/23247096251347403. Epub 2025 Jun 12.
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.
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。