Figueiredo Miguel Pádua, Pato Marco, Amaral Fernando
Department of Orthopaedics, Hospital Prof. Doutor Fernando Fonseca, Estrada IC19, 2720-276 Amadora, Portugal.
J Orthop Case Rep. 2017 Jan-Feb;7(1):75-78. doi: 10.13107/jocr.2250-0685.69.
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory condition. The clinical picture consists of sterile osteomyelitis, typically with multiple-site lesions in the metaphysis of long bones and not uncommonly, symmetrical bone involvement. It is a poorly understood entity, whose prognosis, etiology and ideal treatment are still controversial. The authors report a case of unifocal presentation with an atypical location.
A previously healthy 12-year-old Caucasian girl came to our institution due to progressive pain on her left thigh for the previous 3 months. The initial X-ray showed a permeative, diaphyseal lesion of her left femur, with marked periosteal reaction. The differential initially included Ewing's sarcoma, osteosarcoma, subacute osteomyelitis, and Langerhans cell histiocytosis. Needle and open biopsies demonstrated the presence of chronic inflammatory infiltrate, with fibrosis, but no signs of neoplastic disease. Serologic and microbiological studies failed to demonstrate an infectious etiology. The patient was treated with nonsteroid anti-inflammatories, corticosteroids, and bisphosphonates for 6 months. Although no antibiotics were employed, the patient showed clinical and radiological improvement, at 18-month follow-up.
CRMO is a rare condition, and the absence of specific features constitutes a diagnostic challenge. A high level of suspicion is paramount to avoid unnecessary biopsies and repeated antibiotic regimens. Unifocal presentation of this disease, atypical locations, and absence of recurrence have all been previously reported, with the evidence pointing to a shared etiological process with no distinction being made between these variants. For this reason, the authors believe that the term "nonbacterial osteomyelitis" might be a more all-embracing designation.
慢性复发性多灶性骨髓炎（CRMO）是一种罕见的自身炎症性疾病。临床表现为无菌性骨髓炎，典型表现为长骨干骺端多部位病变，且常为对称性骨受累。这是一种了解甚少的疾病，其预后、病因及理想治疗方法仍存在争议。作者报告一例单灶性且部位不典型的病例。
一名既往健康的12岁白种女孩因左大腿前3个月逐渐加重的疼痛前来我院就诊。最初的X线显示左股骨骨干有渗透性病变，并伴有明显的骨膜反应。鉴别诊断最初包括尤因肉瘤、骨肉瘤、亚急性骨髓炎和朗格汉斯细胞组织细胞增多症。针吸活检和切开活检显示存在慢性炎症浸润及纤维化，但无肿瘤性疾病迹象。血清学和微生物学研究未能证实有感染性病因。患者接受了6个月的非甾体抗炎药、皮质类固醇和双膦酸盐治疗。尽管未使用抗生素，但在18个月的随访中，患者临床和影像学均有改善。
CRMO是一种罕见疾病，缺乏特异性特征构成诊断挑战。高度怀疑对于避免不必要的活检和重复使用抗生素治疗至关重要。此前已有关于本病单灶性表现、非典型部位及无复发的报道，证据表明这些变异型之间存在共同的病因学过程，并无区别。因此，作者认为“非细菌性骨髓炎”这一术语可能是一个更具包容性的称谓。
