Department of Οrthopedics, University Hospital of Ioannina, Stavros Niarchos Avenue 1, Karaoli kai Dimitriou 5, Ioannina, 45332, Greece.
Department of Pathology, University Hospital of Ioannina, Stavros Niarchos Avenue 1, Ioannina, Greece.
Rheumatol Int. 2019 Apr;39(4):751-755. doi: 10.1007/s00296-018-04239-8. Epub 2019 Jan 5.
Idiopathic hip chondrolysis is a rare disorder, the pathophysiology of which has not been fully elucidated. Several theories have been proposed regarding the cause of the disease with some of them involving autoimmune-mediated cartilage destruction. There are several similar features between idiopathic hip chondrolysis and rheumatologic diseases such as juvenile idiopathic arthritis, so whether these two disorders are different or not is still debatable. This case report aims to help comprehending this complex disorder by presenting a case of idiopathic hip chondrolysis with apparent risk factors, such as repetitive microtrauma and presence of HLA-B27 antigens. A 15-year-old HLA-B27 positive male presented with idiopathic hip chondrolysis after excessive walking. Initial treatment consisted of medications including corticosteroids, protected weight bearing and surgical soft tissue release. After failure of all these modalities in restoring the decreased range of motion of the hip, a course of a TNF-inhibitor, etanercept was tried. Alleviation of pain achieved early in the treatment period, but range of motion remained mainly unchanged. Although there was a brief improvement of stiffness for a short period after surgery which lasted for about 3 months, stiffness came back afterwards. Administration of a TNF inhibitor in the following period significantly improved his range of motion. The presence of laboratory findings indicating an autoimmune tendency in this patient supports the hypothesis of susceptibility of these patients to autoimmune reactions, while excessive walking was an apparent trigger factor. In future, traditional treatments may be abandoned in favor of novel medications targeting immunologic pathways.
特发性髋关节软骨溶解症是一种罕见的疾病，其病理生理学尚未完全阐明。关于该病的病因提出了几种理论，其中一些涉及自身免疫介导的软骨破坏。特发性髋关节软骨溶解症和风湿性疾病（如幼年特发性关节炎）有几个相似的特征，因此这两种疾病是否不同仍存在争议。本病例报告旨在通过报告一例有明显危险因素（如反复微创伤和 HLA-B27 抗原存在）的特发性髋关节软骨溶解症来帮助理解这种复杂的疾病。一名 15 岁 HLA-B27 阳性男性在过度行走后出现特发性髋关节软骨溶解症。初始治疗包括药物治疗，包括皮质类固醇、保护承重和手术软组织松解。在所有这些方法都未能恢复髋关节活动度降低的情况下，尝试了一种 TNF 抑制剂，依那西普。在治疗早期疼痛得到缓解，但活动度主要没有变化。尽管手术后关节僵硬有短暂改善，但持续了大约 3 个月，随后又恢复了僵硬。在随后的时期使用 TNF 抑制剂显著改善了他的活动度。该患者存在实验室检查结果表明存在自身免疫倾向，支持这些患者易发生自身免疫反应的假说，而过度行走是明显的触发因素。未来，传统治疗方法可能会被放弃，转而采用针对免疫途径的新型药物。
