Department of Neurology.
Department of Clinical Neuroscience and Therapeutics, Graduate School of Biomedical and Health Sciences, Hiroshima University, Higashihiroshima, Hiroshima, Japan.
J Neuropathol Exp Neurol. 2020 Apr 1;79(4):448-457. doi: 10.1093/jnen/nlaa006.
Anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR) and antisignal recognition particle (SRP) antibodies are frequently associated with immune-mediated necrotizing myopathy (IMNM). However, the difference in clinical manifestations between anti-HMGCR and anti-SRP antibodies is unclear. HMGCR is an essential enzyme for cholesterol biosynthesis and is inhibited by statins that regulate apoptosis of Bcl-2-positive and beta chemokine receptor 4 (CCR4)-positive lymphoma cells. In this study, we aimed to clarify Bcl-2 and CCR4 expressions of lymphocytes in anti-HMGCR antibody-positive IMNM and explore the difference between anti-HMGCR antibody-positive myopathy and other inflammatory myopathies. We retrospectively examined Bcl-2- and CCR4-positive lymphocyte infiltrations in muscle and skin biopsy specimens from 19 anti-HMGCR antibody-positive patients and 75 other idiopathic inflammatory myopathies (IIMs) patients. A higher incidence of Bcl-2- and CCR4-positive lymphocytes was detected in the muscle and skin of anti-HMGCR antibody-positive IMNM patients (p < 0.001). In 5 patients with anti-HMGCR antibodies, Bcl-2-positive lymphocytes formed lymphocytic accumulations, which were not observed in other IIMs. Low-density lipoprotein cholesterol levels were not increased except for patients with Bcl-2-positive lymphocytic accumulations (p = 0.010). Bcl-2 and CCR4 lymphocyte infiltrations could be a pathological characteristic of anti-HMGCR antibody-positive IMNM.
抗 3-羟-3-甲基戊二酰辅酶 A 还原酶 (HMGCR) 和抗信号识别颗粒 (SRP) 抗体常与免疫介导的坏死性肌病 (IMNM) 相关。然而,抗 HMGCR 抗体和抗 SRP 抗体之间的临床表现差异尚不清楚。HMGCR 是胆固醇生物合成的必需酶,被他汀类药物抑制,他汀类药物调节 Bcl-2 阳性和β趋化因子受体 4 (CCR4) 阳性淋巴瘤细胞的凋亡。在这项研究中,我们旨在阐明抗 HMGCR 抗体阳性 IMNM 中淋巴细胞的 Bcl-2 和 CCR4 表达,并探讨抗 HMGCR 抗体阳性肌病与其他炎症性肌病之间的差异。我们回顾性地检查了 19 例抗 HMGCR 抗体阳性患者和 75 例其他特发性炎症性肌病 (IIM) 患者的肌肉和皮肤活检标本中 Bcl-2-和 CCR4-阳性淋巴细胞浸润。抗 HMGCR 抗体阳性 IMNM 患者的肌肉和皮肤中 Bcl-2-和 CCR4-阳性淋巴细胞的发生率更高(p<0.001)。在 5 例抗 HMGCR 抗体阳性患者中,Bcl-2 阳性淋巴细胞形成淋巴细胞聚集,而在其他 IIM 中未观察到。除了 Bcl-2 阳性淋巴细胞聚集的患者外(p=0.010),低密度脂蛋白胆固醇水平没有升高。Bcl-2 和 CCR4 淋巴细胞浸润可能是抗 HMGCR 抗体阳性 IMNM 的病理特征。