关键词: CRMO Chronic non-bacterial osteomyelitis SAPHO-syndrome Spondyloarthritis

Mesh : Acquired Hyperostosis Syndrome / diagnosis drug therapy Adolescent Adult Age of Onset Anti-Inflammatory Agents, Non-Steroidal / therapeutic use Antirheumatic Agents / therapeutic use Child Female Humans Magnetic Resonance Imaging Male Methotrexate / therapeutic use Middle Aged Osteomyelitis / diagnosis drug therapy Retrospective Studies Steroids / therapeutic use Time-to-Treatment Tomography, X-Ray Computed Treatment Outcome Young Adult

来  源:   DOI:10.1186/s12969-019-0353-2   PDF(Sci-hub)   PDF(Pubmed)

Abstract:
BACKGROUND: To compare clinical presentation, diagnostic and treatment strategies, and outcome between pediatric and adult patients with chronic non-bacterial osteomyelitis (CNO).
METHODS: Retrospective single-centre comparative study of pediatric and adult patients diagnosed with chronic recurrent multifocal osteomyelitis (CRMO)/CNO or synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome treated at the Medical University of Graz.
RESULTS: 24 pediatric patients diagnosed with CRMO/CNO and 10 adult patients diagnosed with SAPHO syndrome were compared. Median age at diagnosis was 12.3 years (range 7.9-18.9) in the pediatric group and 32.5 years (range 22-56) in the adult group. Median time to diagnosis was shorter in children than in adults (0.3 vs. 1.0 years). Initial clinical presentation, laboratory and histopathological findings were similar in children and adults. Mean numbers of bone lesions were comparable between pediatric and adult patients (3.1 vs. 3.0), as were rates of skin involvement (33% vs. 30%). Sternal involvement was more frequent in adults whereas involvement of clavicle and long bones was more frequent in children (41.7% vs.10, 33% vs. 10%). Computerized tomography (CT) was used more often in adults, whereas whole-body magnetic resonance imaging (MRI) was used only in children. Bisphosphonates were applied more often in children and outcome was better in children than in adults (62.5% vs.30%).
CONCLUSIONS: Results of our study suggest that CNO/CRMO and SAPHO syndrome in children and adults might represent a single clinical syndrome that needs a similar diagnostic and therapeutic approach.
摘要:
背景:为了比较临床表现,诊断和治疗策略,儿童和成人慢性非细菌性骨髓炎(CNO)患者的结局。
方法:诊断为慢性复发性多灶性骨髓炎(CRMO)/CNO或滑膜炎的儿童和成人患者的回顾性单中心比较研究,痤疮,脓疱病,骨增生,和在格拉茨医科大学治疗的骨炎(SAPHO)综合征。
结果:比较了24例诊断为CRMO/CNO的儿科患者和10例诊断为SAPHO综合征的成人患者。诊断时的中位年龄在儿科组为12.3岁(范围7.9-18.9),在成人组为32.5岁(范围22-56)。儿童的中位诊断时间短于成人(0.3vs.1.0年)。最初的临床表现,儿童和成人的实验室和组织病理学结果相似.儿童和成人患者的平均骨病变数量相当(3.1vs.3.0),皮肤受累率(33%vs.30%)。成人的内脏受累更为频繁,而儿童的锁骨和长骨受累更为频繁(41.7%vs.10,33%vs.10%)。计算机断层扫描(CT)更常用于成人,而全身磁共振成像(MRI)仅用于儿童。双膦酸盐在儿童中的应用频率更高,儿童的结果优于成人(62.5%vs.30%)。
结论:我们的研究结果表明,儿童和成人的CNO/CRMO和SAPHO综合征可能代表一种单一的临床综合征,需要类似的诊断和治疗方法。
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