Qi Zhou, Liu Jianli, Li Guoqiang, Zhang Yinian
Department of Neurosurgery, Lanzhou University Second Hospital, Lanzhou, China.
Department of Medical Imaging, Lanzhou University Second Hospital, Lanzhou, China.
Front Neurol. 2022 Jun 21;13:878414. doi: 10.3389/fneur.2022.878414. eCollection 2022.
Immunoglobulin G4-related disease (IgG4-RD) is an autoimmune disease that affects several organs. An inflammatory pseudotumor is a histologically proven benign tumor-like lesion that most commonly involves the lung and orbit. It is rare in the central nervous system, but rarest in the spinal canal. In this report, we present a case of IgG4-related intramedullary spinal inflammatory pseudotumor, along with a literature review. A 29-year-old male was transferred to the Department of Neurosurgery of Lanzhou University Second Hospital with progressive quadriparesis after numbness and weakness in both lower limbs for 50 days. Enhanced magnetic resonance imaging (MRI) of the spine revealed an isointense signal on T1-weighted images and a hyperintense signal on T2-weighted images from an enhanced mass located at the thoracic vertebrae region, for which a schwannoma was highly suspected. Then, a posterior median approach was adopted. The lesion was resected. The patient received further glucocorticoid after the diagnosis of an IgG4-related inflammatory pseudotumor was established, and the patient's symptoms improved, such as quadriparesis and lower limb weakness. This case highlights the importance of considering IgG4-related inflammatory pseudotumor as a differential diagnosis in patients with lesions involving the spinal intramedullary compartment and lower limb weakness when other more threatening causes have been excluded. IgG4-related inflammatory pseudotumor is etiologically unclear and prognostically unpredictable, and imaging may not help establish the diagnosis of IgG4-related inflammatory pseudotumor due to its resemblance to malignant tumors, and total resection might not be warranted. Glucocorticoid and surgery are usually the first-line treatments used.
免疫球蛋白G4相关性疾病(IgG4-RD)是一种累及多个器官的自身免疫性疾病。炎性假瘤是一种经组织学证实的良性肿瘤样病变,最常累及肺和眼眶。它在中枢神经系统中罕见,而在椎管中最为罕见。在本报告中,我们介绍了一例IgG4相关性脊髓内炎性假瘤病例,并进行文献复习。一名29岁男性因双下肢麻木无力50天后出现进行性四肢瘫,被转至兰州大学第二医院神经外科。脊柱增强磁共振成像(MRI)显示,位于胸椎区域的强化肿块在T1加权图像上呈等信号,在T2加权图像上呈高信号,高度怀疑为神经鞘瘤。随后,采用后正中入路。切除病变。在确诊为IgG4相关性炎性假瘤后,患者接受了进一步的糖皮质激素治疗,其症状有所改善,如四肢瘫和下肢无力。该病例强调了在排除其他更具威胁性的病因后,对于累及脊髓髓内腔且伴有下肢无力的病变患者,将IgG4相关性炎性假瘤作为鉴别诊断的重要性。IgG4相关性炎性假瘤病因不明,预后难以预测,由于其与恶性肿瘤相似,影像学检查可能无助于IgG4相关性炎性假瘤的诊断,且可能无需进行全切。糖皮质激素和手术通常是一线治疗方法。