Department of Rheumatology, National Reference Center for Rare Autoimmune Diseases (RESO), INSERM UMR-S 1109, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
National Referral Centre for Rare Autoimmune and Systemic Diseases, Department of Internal Medicine, Hôpital Cochin, AP-HP Centre, Université Paris Cité, Paris, France.
Nat Rev Rheumatol. 2024 Jun;20(6):347-360. doi: 10.1038/s41584-024-01113-9. Epub 2024 May 2.
Relapsing polychondritis is a rare inflammatory disease characterized by recurrent inflammation of cartilaginous structures, mainly of the ears, nose and respiratory tract, with a broad spectrum of accompanying systemic features. Despite its rarity, prompt recognition and accurate diagnosis of relapsing polychondritis is crucial for appropriate management and optimal outcomes. Our understanding of relapsing polychondritis has changed markedly in the past couple of years with the identification of three distinct patient clusters that have different clinical manifestations and prognostic outcomes. With the progress of pangenomic sequencing and the discovery of new somatic and monogenic autoinflammatory diseases, new differential diagnoses have emerged, notably the vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome, autoinflammatory diseases and immune checkpoint inhibitor-related adverse events. In this Review, we present a detailed update of the newly identified clusters and highlight red flags that should raise suspicion of these alternative diagnoses. The identification of these different clusters and mimickers has a direct impact on the management, follow-up and prognosis of patients with relapsing polychondritis and autoinflammatory syndromes.
复发性多软骨炎是一种罕见的炎症性疾病,其特征为软骨结构反复发生炎症,主要累及耳部、鼻部和呼吸道,同时伴有广泛的伴随全身特征。尽管其发病率较低,但及时识别和准确诊断复发性多软骨炎对于进行适当的管理和获得最佳结果至关重要。在过去几年中,我们对复发性多软骨炎的认识发生了显著变化,确定了三个不同的患者群体,这些群体具有不同的临床表现和预后结果。随着全基因组测序的进展和新的体细胞和单基因自身炎症性疾病的发现,出现了新的鉴别诊断,尤其是空泡、E1 酶、X 连锁、自身炎症、体细胞(VEXAS)综合征、自身炎症性疾病和免疫检查点抑制剂相关不良事件。在这篇综述中,我们详细介绍了新确定的患者群体,并强调了应引起对这些替代诊断怀疑的警示信号。这些不同群体和类似疾病的确定对复发性多软骨炎和自身炎症性综合征患者的管理、随访和预后有直接影响。