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一例罕见的重叠性免疫球蛋白G4相关疾病与系统性红斑狼疮病例

An Unusual Case of Overlapping Immunoglobulin G4-Related Disease and Systemic Lupus Erythematosus.

作者信息

Burillo Simões Pilar, Martins João, Do Mar Menezes Maria, Sousa João, Jorge Cristina

机构信息

Nephrology, Unidade Local de Saúde de São José, Lisbon, PRT.

出版信息

Cureus. 2024 Dec 9;16(12):e75362. doi: 10.7759/cureus.75362. eCollection 2024 Dec.

Abstract

Immunoglobulin G4-related disease (IgG4-RD) and systemic lupus erythematosus (SLE) are multisystemic autoimmune disorders that can present with renal manifestations. Overlapping cases of these diseases are extremely rare and present both diagnostic and therapeutic challenges. We report the case of a 70-year-old male with a history of autoimmune pancreatitis, who was admitted with fatigue, weight loss, and worsening kidney function. Laboratory tests revealed anemia with a positive Coombs test, leucopenia, elevated IgG4, hypocomplementemia, and positive results for ANA, anti-double-stranded DNA (dsDNA), anti-nucleosome, anti-RP11 antibodies, and rheumatoid factor. A spot urine sample showed subnephrotic proteinuria without hematuria. The patient met the criteria for both SLE and possible IgG4-RD, but the cause of the worsening renal function remained unclear, prompting a kidney biopsy. The biopsy revealed a lymphoplasmacytic infiltrate, storiform fibrosis, and IgG4-positive staining, consistent with IgG4-related tubulointerstitial nephritis, but without evidence of lupus nephritis. The patient was treated with prednisolone, resulting in improvement of both his symptoms and kidney function. However, significant leukopenia, anemia, and elevated anti-dsDNA titers persisted, which were presumed to be secondary to the overlapping SLE. Hydroxychloroquine and azathioprine were added to the treatment regimen, leading to improvement in cytopenias at the three-month follow-up. This case underscores the importance of kidney biopsy in suspected overlapping autoimmune diseases for identifying kidney involvement and guiding treatment, although evidence regarding optimal therapy remains limited.

摘要

免疫球蛋白G4相关疾病(IgG4-RD)和系统性红斑狼疮(SLE)是可出现肾脏表现的多系统自身免疫性疾病。这些疾病的重叠病例极为罕见,带来了诊断和治疗方面的挑战。我们报告一例70岁男性患者,有自身免疫性胰腺炎病史,因疲劳、体重减轻和肾功能恶化入院。实验室检查显示贫血且库姆斯试验阳性、白细胞减少、IgG4升高、补体血症、抗核抗体(ANA)、抗双链DNA(dsDNA)、抗核小体、抗RP11抗体及类风湿因子检测结果均为阳性。一份随机尿样显示为亚肾病范围蛋白尿,无血尿。该患者符合SLE和可能的IgG4-RD的标准,但肾功能恶化的原因仍不清楚,因此进行了肾活检。活检显示淋巴细胞和浆细胞浸润、席纹状纤维化及IgG4阳性染色,符合IgG4相关肾小管间质性肾炎,但无狼疮性肾炎证据。患者接受泼尼松龙治疗后,症状和肾功能均有改善。然而,严重的白细胞减少、贫血及抗dsDNA滴度升高持续存在,推测这是由于重叠的SLE所致。治疗方案中加用了羟氯喹和硫唑嘌呤,在三个月的随访中血细胞减少情况有所改善。该病例强调了在疑似自身免疫性疾病重叠时肾活检对于确定肾脏受累情况及指导治疗的重要性,尽管关于最佳治疗的证据仍然有限。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1bd3/11710871/e1fac203142f/cureus-0016-00000075362-i01.jpg

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