Berkowitz Y J, Greenwood S J, Cribb G, Davies K, Cassar-Pullicino V N
Department of Diagnostic Imaging, Robert Jones and Agnes Hunt Orthopaedic Hospital NHS Foundation Trust, Oswestry, Shropshire, SY10 7AG, UK.
Department of Orthopaedic Oncology, Robert Jones and Agnes Hunt Orthopaedic Hospital NHS Foundation Trust, Oswestry, Shropshire, SY10 7AG, UK.
Skeletal Radiol. 2018 Apr;47(4):563-568. doi: 10.1007/s00256-017-2812-5. Epub 2017 Nov 9.
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare condition thought to be under-diagnosed, with a true prevalence of more than the 1 in 10,000 estimated. It is a condition that is classically described as polyostotic with a relapsing and remitting course, preferentially affecting the metaphyses of tubular bones in the pediatric population. Lesions have characteristic appearances of cortical hyperostosis and mixed lytic/sclerotic medullary appearances radiographically, with active osteitis and periostitis best seen with fluid-sensitive sequences on magnetic resonance imaging (MRI). There are reports of lesions resolving on follow-up radiographs and MRI scans, but no supporting images. In particular, although the marrow appearances and degree of osteitis have been shown to improve on MRI, complete resolution and remodeling back to normal has never been demonstrated. We present a case of a lesion that has completely healed and remodeled back to normal appearances on both radiographs and MRI, and consider this the standard for the often loosely used terms "normalization" and "resolution". We discuss the implications of this for our understanding of the natural history of CRMO, and how this adds weight to the condition being significantly under-diagnosed. It provides a "gold standard" to be aimed for when assessing treatments for CRMO, and the optimal outcomes that are possible. It also provides further insight into the potential of pediatric bone to recover and remodel when affected by inflammatory conditions.
慢性复发性多灶性骨髓炎(CRMO)是一种罕见疾病,据认为诊断不足,实际患病率超过万分之一。该病典型表现为多骨受累,病程呈复发缓解型,在儿童人群中优先累及管状骨的干骺端。影像学上,病变具有皮质骨增生以及骨髓呈溶骨/硬化混合表现的特征,磁共振成像(MRI)上的液体敏感序列最能清晰显示活动性骨炎和骨膜炎。有报道称病变在随访X线片和MRI扫描时有所缓解,但无相关支持图像。特别是,尽管MRI显示骨髓表现和骨炎程度有所改善,但从未证实病变能完全消退并重塑恢复正常。我们报告一例病变在X线片和MRI上均完全愈合并重塑恢复正常表现的病例,并将此视为常用于描述“正常化”和“消退”的标准。我们讨论了这对我们理解CRMO自然病史的意义,以及这如何进一步表明该病存在显著诊断不足的情况。它为评估CRMO治疗方法及可能达到的最佳疗效提供了一个可为之努力的“金标准”。它还进一步揭示了儿童骨骼在受到炎症影响时恢复和重塑的潜力。