Highstein Mallory Jayne, Mallen Jonathan, Tham Tristan, Brennan Tara, Boubour Alexandra, Opher Elena, Wolf Vira, Singh Prabhjyot, Costantino Peter
Hofstra North Shore, LIJ School of Medicine, Hempstead, New York, United States.
New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States.
J Neurol Surg Rep. 2015 Nov;76(2):e265-9. doi: 10.1055/s-0035-1564602. Epub 2015 Oct 9.
Introduction Immunoglobulin subtype G4-related disease (IgG4-RD) is a fibroinflammatory disease of unknown etiology, with manifestations involving nearly every organ system. Its association with foreign bodies is not established. Here, we present a novel case of IgG4-RD in response to foreign body injection. Case Description A 58-year-old woman presented with history of persistent left facial pain, xerophthalmia, blurred vision, and trismus. The patient's medical history was significant for left-sided temporomandibular joint (TMJ) reconstruction with silicone injection into the joint. Magnetic resonance imaging revealed a lesion in the left skull base. Biopsies demonstrated the cardinal histopathological features of IgG4-RD. The patient was treated with a tapering dose of prednisolone followed by rituximab, resulting in tumor shrinkage and resolution of her symptoms. Discussion This is the first reported case of IgG4-RD potentially precipitated by a foreign body, in this case injected silicone into the TMJ. The pathogenesis and etiology of IgG4-RD is still not fully elucidated, but allergic and reactive inflammatory reactions have been implicated in the disease process. This case report should raise the idea of reactive foreign bodies as a causative agent for IgG4-RD.
引言 免疫球蛋白G4相关疾病(IgG4-RD)是一种病因不明的纤维炎性疾病,其表现累及几乎每个器官系统。其与异物的关联尚未确立。在此,我们报告一例因异物注射引发的IgG4-RD新病例。病例描述 一名58岁女性,有持续性左面部疼痛、干眼症、视力模糊和牙关紧闭病史。患者有左侧颞下颌关节(TMJ)用硅胶注射进行重建的病史。磁共振成像显示左侧颅底有一个病变。活检显示了IgG4-RD的主要组织病理学特征。患者接受了逐渐减量的泼尼松龙治疗,随后使用利妥昔单抗,肿瘤缩小且症状缓解。讨论 这是首例报道的可能由异物引发的IgG4-RD病例,在本病例中是向TMJ注射硅胶。IgG4-RD的发病机制和病因仍未完全阐明,但过敏和反应性炎症反应已被认为与疾病过程有关。本病例报告应引发人们将反应性异物视为IgG4-RD致病因素的思考。