Department of Nephrology, Teine Keijinkai Medical Center, Sapporo, Hokkaido, Japan.
Hokkaido Renal Pathology Center, Sapporo, Hokkaido, Japan.
CEN Case Rep. 2021 Aug;10(3):435-441. doi: 10.1007/s13730-021-00583-0. Epub 2021 Feb 22.
Selective immunoglobulin M deficiency (SIgMD) is the isolated absence of serum immunoglobulin M (IgM) with normal levels of other serum immunoglobulins. SIgMD is associated with infections and autoimmune diseases. While there are few reports on SIgMD complicated by systemic lupus erythematosus (SLE), there are no reports on SIgMD complicated by SLE and antiphospholipid syndrome (APS); we present the first report of this kind. A 61-year-old Japanese woman presented with microscopic hematuria and proteinuria. Clinical investigations revealed an elevated serum creatinine level, an undetectable serum IgM level, and seropositivity of antinuclear antibody, anti-Smith antibody, and double-stranded DNA antibody. Radiological investigations were unremarkable. Renal biopsy revealed focal and segmental mesangial cell proliferation; thickened glomerular capillary walls; and IgG, IgA, C3, and C1q deposition, which indicated class III (A/C) lupus nephritis (Renal Pathology Society/International Society of Nephrology classification). Furthermore, anti-CLβ2GP1 antibody positivity and deep vein thrombosis were noted, which fulfilled the revised Sapporo classification criteria for the diagnosis of APS. Thus, she was diagnosed with SIgMD complicated by SLE and APS. The patient was treated with prednisolone, mycophenolate mofetil, and warfarin. After a 1-year follow-up, she achieved clinical remission of SLE and APS without infectious complications; however, the serum IgM level remained undetectable. In conclusion, SIgMD can be complicated by autoimmune disorders. Although rare, we recommend that SLE and APS be considered in patients with SIgMD who present with hematuria, proteinuria, and deep vein thrombosis. We also recommend measuring the titers of antinuclear antibodies, double-stranded DNA antibodies, and anti-CLβ2GP1 antibodies.
选择性免疫球蛋白 M 缺乏症(SIgMD)是指血清免疫球蛋白 M(IgM)水平正常,但其他血清免疫球蛋白缺失的疾病。SIgMD 与感染和自身免疫性疾病有关。虽然有少数关于 SIgMD 并发系统性红斑狼疮(SLE)的报道,但没有关于 SIgMD 并发 SLE 和抗磷脂综合征(APS)的报道;我们报告了首例此类病例。一名 61 岁的日本女性因镜下血尿和蛋白尿就诊。临床检查发现血清肌酐水平升高、血清 IgM 水平无法检测、抗核抗体、抗 Smith 抗体和双链 DNA 抗体阳性。影像学检查无明显异常。肾活检显示局灶性和节段性系膜细胞增殖;肾小球毛细血管壁增厚;以及 IgG、IgA、C3 和 C1q 沉积,提示为 III 级(A/C)狼疮性肾炎(肾脏病理学会/国际肾脏病学会分类)。此外,还发现抗 CLβ2GP1 抗体阳性和深静脉血栓形成,符合 APS 的修订版 Sapporo 分类标准。因此,诊断为 SIgMD 并发 SLE 和 APS。患者接受泼尼松龙、霉酚酸酯和华法林治疗。经过 1 年的随访,SLE 和 APS 达到临床缓解,无感染并发症;然而,血清 IgM 水平仍无法检测到。总之,SIgMD 可并发自身免疫性疾病。虽然罕见,但我们建议对出现血尿、蛋白尿和深静脉血栓形成的 SIgMD 患者考虑 SLE 和 APS。我们还建议测量抗核抗体、双链 DNA 抗体和抗 CLβ2GP1 抗体的滴度。