Haşlak Fatih, Akay Nergis, Gül Ümit, Günalp Aybüke, Kılıç Könte Elif, Şahin Sezgin, Kasapçopur Özgür
Department of Pediatric Rheumatology, İstanbul University-Cerrahpaşa, Cerrahpaşa Faculty of Medicine, İstanbul, Türkiye.
Balkan Med J. 2025 Jan 2;42(1):5-13. doi: 10.4274/balkanmedj.galenos.2024.2024-11-129.
Autoinflammatory bone diseases (AIBDs) constitute a recently identified subset of autoinflammatory diseases. These conditions are characterized by an exaggerated inflammatory response in the bones without any apparent etiology. Inflammatory bone lesions associated with AIBDs exhibit chronic inflammation, are typically culture-negative, and do not exhibit discernible microorganisms on histopathological examination. The most common and representative AIBD is chronic non-bacterial osteomyelitis (CNO), which is also known as chronic recurrent multifocal osteomyelitis. Another variant of CNO, which is typically observed in older teenagers or adults, is known as synovitis, acne, hyperostosis, pustulosis, osteitis syndrome. This condition is distinguished by its notable skin manifestations. Advancements in genetic research have led to the identification of three novel monogenic subtypes within the category of AIBDs. These include Majeed syndrome, pyogenic arthritis, pyoderma gangrenosum, and acne syndrome, and interleukin-1 receptor antagonist deficiency syndrome. Another monogenic AIBD, called cherubism, affects only the maxilla and mandible. Data on the diagnosis and treatment of these rare diseases are extremely limited. However, if not diagnosed and treated promptly, it can result in significant complications, including severe disability and mortality. Thus, it is imperative to maintain a high level of clinical awareness of these diseases. These rare diagnoses should be considered in patients with musculoskeletal complaints in whom no specific etiology can be identified or in patients with systemic manifestations such as cutaneous and gastrointestinal symptoms or fever. In such patients, the diagnostic process, which encompasses imaging and genetic studies, should be initiated promptly.
自身炎症性骨病(AIBDs)是最近确定的自身炎症性疾病的一个子集。这些病症的特征是在没有任何明显病因的情况下,骨骼中出现过度的炎症反应。与AIBDs相关的炎症性骨病变表现为慢性炎症,通常培养阴性,并且在组织病理学检查中未显示可辨别的微生物。最常见且具有代表性的AIBD是慢性非细菌性骨髓炎(CNO),也称为慢性复发性多灶性骨髓炎。CNO的另一种变体,通常在青少年或成年人中观察到,称为滑膜炎、痤疮、骨肥厚、脓疱病、骨炎综合征。这种病症以其明显的皮肤表现为特征。基因研究的进展导致在AIBDs类别中鉴定出三种新的单基因亚型。这些包括马吉德综合征、化脓性关节炎、坏疽性脓皮病和痤疮综合征以及白细胞介素-1受体拮抗剂缺乏综合征。另一种单基因AIBD,称为颌骨肥大症,仅影响上颌骨和下颌骨。关于这些罕见疾病的诊断和治疗的数据极其有限。然而,如果不及时诊断和治疗,可能会导致严重的并发症,包括严重残疾和死亡。因此,必须对这些疾病保持高度的临床认识。对于无法确定具体病因的肌肉骨骼疾病患者或有皮肤和胃肠道症状或发热等全身表现的患者,应考虑这些罕见诊断。在此类患者中,应立即启动包括影像学和基因研究在内的诊断过程。