Case report: Child chronic nonbacterial osteomyelitis with rapid progressive scoliosis-an association with disease?

BackgroundChronic nonbacterial osteomyelitis (CNO) is an auto-inflammatory bone disease that usually develops in childhood. Spinal involvement is a common manifestation of CNO, but it is rare for CNO to lead to rapid progression of scoliosis deformity. Here we present a 9-year-old girl with acute sc...

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Main Authors: Xiaojun Shi (Author), Xiujuan Hou (Author), Haiqin Hua (Author), Xia Dong (Author), Xiaoping Liu (Author), Fengjiao Cao (Author), Chen Li (Author)
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Published: Frontiers Media S.A., 2023-03-01T00:00:00Z.
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001 doaj_ba0a9dedf61d45d98c3364abfc3a40aa
042 |a dc 
100 1 0 |a Xiaojun Shi  |e author 
700 1 0 |a Xiujuan Hou  |e author 
700 1 0 |a Haiqin Hua  |e author 
700 1 0 |a Xia Dong  |e author 
700 1 0 |a Xiaoping Liu  |e author 
700 1 0 |a Fengjiao Cao  |e author 
700 1 0 |a Chen Li  |e author 
245 0 0 |a Case report: Child chronic nonbacterial osteomyelitis with rapid progressive scoliosis-an association with disease? 
260 |b Frontiers Media S.A.,   |c 2023-03-01T00:00:00Z. 
500 |a 2296-2360 
500 |a 10.3389/fped.2023.1076443 
520 |a BackgroundChronic nonbacterial osteomyelitis (CNO) is an auto-inflammatory bone disease that usually develops in childhood. Spinal involvement is a common manifestation of CNO, but it is rare for CNO to lead to rapid progression of scoliosis deformity. Here we present a 9-year-old girl with acute scoliosis with CNO and scoliosis progressed rapidly in 2 months.Case PresentationA 9-year-old girl presented bilateral shoulder inequality with pain in the left hypochondrium for 2 months. Standing spinal x-rays showed right convex scoliosis with a 25° Cobb angle. Chest magnetic resonance imaging (MRI) showed that the T8 vertebra was flattened and local bone was destroyed with bone marrow edema. The bone biopsy showed evidence of fibrosis and chronic inflammatory changes with no specific diagnosis. One month later, her scoliosis and bone destruction deteriorated obviously. Thoracic vertebra MRI showed that the T8 vertebra had a compression fracture. 99mTc-MDP whole-body bone scintigraphy showed intense uptake at T8/9 and the right sacroiliac joint. She was diagnosed with CNO accompanied by rapidly progressive scoliosis. The scoliosis was successfully treated with adalimumab and zoledronic acid, which showed significant improvement after 6 months of follow-up.ConclusionZoledronic acid and adalimumab successfully treated CNO with rapidly progressive scoliosis, but could not prevent vertebral compression. 
546 |a EN 
690 |a scoliosis 
690 |a children 
690 |a autoimmune disease 
690 |a rapid progress 
690 |a chronic nonbacterial osteomyelitis 
690 |a Adalimumab 
690 |a Pediatrics 
690 |a RJ1-570 
655 7 |a article  |2 local 
786 0 |n Frontiers in Pediatrics, Vol 11 (2023) 
787 0 |n https://www.frontiersin.org/articles/10.3389/fped.2023.1076443/full 
787 0 |n https://doaj.org/toc/2296-2360 
856 4 1 |u https://doaj.org/article/ba0a9dedf61d45d98c3364abfc3a40aa  |z Connect to this object online.