Astouati Quentin, Machet Thomas, Houssais Camille, Noury Jean-Baptiste, Allenbach Yves, Gallay Laure, Quere Baptiste, Assan Florence, Benveniste Olivier, Broner Jonathan, Duffau Pierre, Espitia Alexandra, Grasland Anne, Hayem Gilles, Le Guern Véronique, Martis Nihal, Mariampillai Kuberaka, Nocturne Gaëtane, Mariette Xavier, Meyer Alain, Mulleman Denis, Devauchelle-Pensec Valérie, Collet Aurore, Launay David, Hachulla Eric, Cornec Divi, Guellec Dewi, Sanges Sébastien
Département de Médecine Interne et Immunologie Clinique, CHU Lille, Lille, France.
Centre National de Référence Maladies Auto-immunes Systémiques Rares du Nord, Nord-Ouest, Méditerranée et Guadeloupe (CeRAINOM), Lille, France.
Rheumatology (Oxford). 2025 Mar 1;64(3):1431-1436. doi: 10.1093/rheumatology/keae129.
To describe the characteristics of patients with Sjögren's disease (SjD) and inclusion-body myositis (IBM), and how they compare to SjD patients with other inflammatory myopathies (IM).
Patients were retrospectively recruited from 13 French centres and included if they met the ACR/EULAR criteria for SjD and for IM. They were categorized as SjD-IBM if sub-criteria for IBM were met, or as SjD-other IM if not.
SjD-IBM patients (n = 22) were mostly females (86%), with a median [Q1; Q3] age of 54 [38.5; 64] years at SjD diagnosis, and 62 [46.5; 70] years at first IBM symptoms. Although most patients displayed glandular and immunological abnormalities, additional extra-glandular manifestations were uncommon, resulting in moderate disease activity at SjD diagnosis (ESSDAI 5.5 [1; 7.8]). Classic IBM features were frequent, such as progressive symptom onset (59%), asymmetrical (27%) and distal (32%) involvements, dysphagia (41%), low CPK (386.5 [221.8; 670.5] UI/l) and CRP (3.0 [3; 8.5] mg/l) levels. Immunosuppressants were reported as efficient in 55% of cases. Compared with SjD-IBM patients, SjD patients with other IM (n = 50) were significantly younger, displayed more frequent additional extra-glandular disease, higher ESSDAI score (11 [3; 30]), shorter delay between SjD diagnosis and myositis onset (0 [-0.5; 26]), more frequent CPK values over 1000 UI/l (36%), and less frequent classic IBM features.
IBM can occur in SjD patients, with muscle features reminiscent of classic sporadic IBM characteristics, but mostly affecting women. In SjD patients with muscle involvement, extra-glandular manifestations, high ESSDAI score, elevated CPK values and shorter delay after SjD diagnosis plead against IBM.
描述干燥综合征(SjD)合并包涵体肌炎(IBM)患者的特征,以及与其他炎性肌病(IM)的干燥综合征患者相比有何不同。
从13个法国中心回顾性招募患者,若符合美国风湿病学会(ACR)/欧洲抗风湿病联盟(EULAR)关于SjD和IM的标准则纳入研究。若符合IBM的次要标准,则归类为SjD-IBM;若不符合,则归类为SjD-其他IM。
SjD-IBM患者(n = 22)大多为女性(86%),SjD诊断时的年龄中位数[第一四分位数(Q1);第三四分位数(Q3)]为54[38.5;64]岁,首次出现IBM症状时为62[46.5;70]岁。虽然大多数患者有腺体和免疫异常,但额外的腺外表现并不常见,导致SjD诊断时疾病活动度为中度(欧洲干燥综合征疾病活动指数(ESSDAI)5.5[1;7.8])。典型的IBM特征很常见,如症状逐渐出现(59%)、不对称受累(27%)和远端受累(32%)、吞咽困难(41%)、肌酸磷酸激酶(CPK)水平低(386.5[221.8;670.5]国际单位/升)和C反应蛋白(CRP)水平低(3.0[3;8.5]毫克/升)。据报告,55%的病例中免疫抑制剂有效。与SjD-IBM患者相比,其他IM的SjD患者(n = 50)明显更年轻,腺外疾病额外表现更常见,ESSDAI评分更高(11[3;30]),SjD诊断与肌炎发作之间的间隔更短(0[-0.5;26]),CPK值超过1000国际单位/升的情况更常见(36%),典型的IBM特征更少见。
IBM可发生于SjD患者,肌肉特征让人联想到典型的散发性IBM特征,但主要影响女性。在有肌肉受累的SjD患者中,腺外表现、ESSDAI评分高、CPK值升高以及SjD诊断后间隔时间短则不支持IBM的诊断。