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抗合成酶综合征合并抗OJ抗体阳性时的严重手指坏死

Severe finger necrosis in antisynthetase syndrome with positive anti-OJ antibodies.

作者信息

Horiuchi Yugo, Hashimoto Kenichi, Yoshida Ryochi, Sekizawa Akinori, Kubota Akatsuki, Maeda Meiko, Toda Tatsushi, Tanaka Yuji

机构信息

Department of General Medicine National Defense Medical College Tokorozawa Saitama Japan.

Division of Hematology and Rheumatology, Department of Internal Medicine National Defense Medical College Tokorozawa Saitama Japan.

出版信息

Clin Case Rep. 2024 Jun 2;12(6):e8990. doi: 10.1002/ccr3.8990. eCollection 2024 Jun.

Abstract

KEY CLINICAL MESSAGE

In a patient with anti-aminoacyl tRNA synthetase antibody and anti-OJ antibody syndrome, interventions likes warming, prostaglandins, and antiplatelets failed. However, prednisolone pulse treatment rapidly halted disease progression. Patients with mild interstitial pneumonia, myositis, and extremity necrosis should be promptly considered for anti-synthetase syndrome and receive immunosuppression after ruling out other causes.

ABSTRACT

Anti-aminoacyl tRNA synthetase (ARS) autoantibodies are myositis-specific, and patients who test positive for ARS and have common clinical features are usually diagnosed with antisynthetase antibody syndrome (antisynthetase syndrome). Anti-ARS antibodies include histidyl-tRNA synthetase-1 (Jo-1), anti-threonyl (PL-7), anti-alanyl (PL-12), anti-glycyl (EJ), anti-asparaginyl (KS), anti-tyrosyl (Ha), and anti-phenylalanyl (Zo) tRNA synthetases. Among these, anti-isoleucyl tRNA synthetase (OJ) autoantibodies are extremely rare, and patients with these are frequently complicated by interstitial pneumonia. We report the case of an older man with ARS antibody syndrome who tested positive for anti-OJ and anti-Sjögren's-syndrome-related antigen A (Ro-52) antibodies. He had muscle weakness due to myositis and unparalleled rapid and severe finger necrosis. Pulsed prednisolone effectively treated the myositis symptoms and terminated the progression of finger necrosis.

摘要

关键临床信息

对于抗氨酰tRNA合成酶抗体和抗OJ抗体综合征患者,保暖、使用前列腺素和抗血小板等干预措施均无效。然而,泼尼松龙冲击治疗迅速阻止了疾病进展。对于患有轻度间质性肺炎、肌炎和肢体坏死的患者,应及时考虑抗合成酶综合征,并在排除其他病因后接受免疫抑制治疗。

摘要

抗氨酰tRNA合成酶(ARS)自身抗体具有肌炎特异性,ARS检测呈阳性且具有常见临床特征的患者通常被诊断为抗合成酶抗体综合征(抗合成酶综合征)。抗ARS抗体包括组氨酰-tRNA合成酶-1(Jo-1)、抗苏氨酰(PL-7)、抗丙氨酰(PL-12)、抗甘氨酰(EJ)、抗天冬酰胺酰(KS)、抗酪氨酰(Ha)和抗苯丙氨酰(Zo)tRNA合成酶。其中,抗异亮氨酰tRNA合成酶(OJ)自身抗体极为罕见,此类患者常并发间质性肺炎。我们报告了一例老年男性ARS抗体综合征患者,其抗OJ和抗干燥综合征相关抗原A(Ro-52)抗体检测呈阳性。他因肌炎出现肌肉无力,并伴有罕见的快速且严重的手指坏死。泼尼松龙冲击治疗有效缓解了肌炎症状并终止了手指坏死的进展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e11/11144602/90cbc47e56aa/CCR3-12-e8990-g001.jpg

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