National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health (NIH), Bethesda, MD, USA.
Division of Rheumatology, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, USA.
Rheumatology (Oxford). 2021 Feb 1;60(2):829-836. doi: 10.1093/rheumatology/keaa436.
Pneumocystis jirovecii pneumonia (PJP) is associated with significant morbidity and mortality in adult myositis patients; however, there are few studies examining PJP in juvenile myositis [juvenile idiopathic inflammatory myopathy (JIIM)]. The purpose of this study was to determine the risk factors and clinical phenotypes associated with PJP in JIIM.
An research electronic data capture (REDCap) questionnaire regarding myositis features, disease course, medications and PJP infection characteristics was completed by treating physicians for 13 JIIM patients who developed PJP (PJP+) from the USA and Canada. Myositis features and medications were compared with 147 JIIM patients without PJP (PJP-) from similar geographic regions who enrolled in National Institutes of Health natural history studies.
PJP+ patients were more often of Asian ancestry than PJP- patients [odds ratio (OR) 8.7; 95% CI 1.3, 57.9]. Anti- melanoma differentiation associated protein 5 (MDA5) autoantibodies (OR 12.5; 95% CI 3.0, 52.4), digital infarcts (OR 43.8; 95% CI 4.2, 460.2), skin ulcerations (OR 12.0; 95% CI 3.5, 41.2) and interstitial lung disease (OR 10.6; 95% CI 2.1, 53.9) were more frequent in PJP+ patients. Before PJP diagnosis, patients more frequently received pulse steroids, rituximab and more immunosuppressive therapy compared with PJP- patients. Seven PJP+ patients were admitted to the intensive care unit and four patients died due to PJP or its complications.
PJP is a severe infection in JIIM that can be associated with mortality. Having PJP was associated with more immunosuppressive therapy, anti-MDA5 autoantibodies, Asian race and certain clinical features, including digital infarcts, cutaneous ulcerations and interstitial lung disease. Prophylaxis for PJP should be considered in juvenile myositis patients with these features.
肺囊虫肺炎(PJP)与成人皮肌炎患者的发病率和死亡率显著相关;然而,关于少年皮肌炎(JIIM)中 PJP 的研究甚少。本研究旨在确定 JIIM 中与 PJP 相关的危险因素和临床表型。
通过对来自美国和加拿大的 13 例 JIIM 患者进行研究电子数据捕获(REDCap)问卷,调查他们的皮肌炎特征、疾病过程、药物使用情况以及 PJP 感染特征。对这些 JIIM 患者的皮肌炎特征和药物使用情况与来自相似地理区域的、参加美国国立卫生研究院自然史研究的 147 例无 PJP(PJP-)JIIM 患者进行了比较。
PJP+患者的亚洲血统比 PJP-患者更为常见[比值比(OR)8.7;95%可信区间(CI)1.3,57.9]。抗黑色素瘤分化相关蛋白 5(MDA5)自身抗体(OR 12.5;95% CI 3.0,52.4)、指(趾)端梗死(OR 43.8;95% CI 4.2,460.2)、皮肤溃疡(OR 12.0;95% CI 3.5,41.2)和间质性肺病(OR 10.6;95% CI 2.1,53.9)在 PJP+患者中更为常见。在 PJP 诊断之前,与 PJP-患者相比,PJP+患者更频繁地接受脉冲类固醇、利妥昔单抗和更多的免疫抑制治疗。7 例 PJP+患者入住重症监护病房,4 例患者因 PJP 或其并发症死亡。
PJP 是 JIIM 中的一种严重感染,可导致死亡。患有 PJP 与更多的免疫抑制治疗、抗 MDA5 自身抗体、亚洲种族和某些临床特征(包括指(趾)端梗死、皮肤溃疡和间质性肺病)相关。在具有这些特征的少年皮肌炎患者中,应考虑 PJP 的预防。