Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China

Objective  To investigate the clinical features and treatment outcomes of chronic non-bacterial osteomyelitis (CNO) from five tertiary pediatric rheumatology services in China and provide possible treatment options for clinicians.  Methods  In this multicenter, retrospective study, pediatric patient...

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Main Authors: LIU Haimei, ZHANG Tianyu, MA Le, ZHANG Zhiyong, XU Meng, ZHANG Tao, XU Hong, TANG Xuemei, YANG Sirui, YU Haiguo, SONG Hongmei, SUN Li
Format: Article
Language:zho
Published: Editorial Office of Medical Journal of Peking Union Medical College Hospital 2023-03-01
Series:Xiehe Yixue Zazhi
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Online Access:https://xhyxzz.pumch.cn/en/article/doi/10.12290/xhyxzz.2022-0712
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author LIU Haimei
ZHANG Tianyu
MA Le
ZHANG Zhiyong
XU Meng
ZHANG Tao
XU Hong
TANG Xuemei
YANG Sirui
YU Haiguo
SONG Hongmei
SUN Li
author_facet LIU Haimei
ZHANG Tianyu
MA Le
ZHANG Zhiyong
XU Meng
ZHANG Tao
XU Hong
TANG Xuemei
YANG Sirui
YU Haiguo
SONG Hongmei
SUN Li
author_sort LIU Haimei
collection DOAJ
description Objective  To investigate the clinical features and treatment outcomes of chronic non-bacterial osteomyelitis (CNO) from five tertiary pediatric rheumatology services in China and provide possible treatment options for clinicians.  Methods  In this multicenter, retrospective study, pediatric patients diagnosed with CNO in Children's Hospital of Fudan University, Peking Union Medical College Hospital, Children's Hospital of Nanjing Medical University, Children's Hospital of Chongqing Medical University and the First Hospital of Jilin University from March 2014 to August 2022 were included. According to the treatment plan, the patients were divided into nonsteroidal anti-inflammatory drugs (NSAID) group, traditional disease modifying anti-rheumatic drugs (DMARD) group, tumor necrosis factor inhibitor (TNF-i) group, bisphosphonate group, and bisphosphonate+TNF-i group, and physician global assessment score was used to evaluate the efficacy.  Results  Retrospective data analysis showed that the disease was more common in boy sthan girls(31 vs. 27), and the average age at onset of symptoms was 8.5±3.4 years, the mean age of diagnosis was 9.8±3.2 years, and the Median follow-up was 10.9(5.0, 30.1) months. A total of 46 cases (79.3%) were diagnosed in the last 3 years. Bone pain and / or arthralgia was the predominant symptom in 54(93.1%) patients followed by fever in 31(53.4%) patients. Raised inflammatory markers (ESR and CRP) were present in 87.9% and 77.6% of the patients. The most frequently affected bones were lower limb bones, including 42 cases of femur and 41 cases of tibia; 3 patients had compression fractures of the vertebrae at the time of diagnosis. Bone biopsy was conducted in 33 cases, and subacute or chronic osteomyelitis manifested with inflammatory cells infiltration were detected. The remission rate was not statistically different between treatment groups (P=0.562), with the remission rate at 11 months in the bisphosphonate+TNF-i group being 60.0%, 55.6% and 33.3% in the NSAID and DMARD groups respectively; 66.7% and 52.4% in the NSAID and TNF-i groups respectively at 12 months. A total of 40 patients were followed up for ≥6 months, with the remission rate of 81.3%.  Conclusions  Our multicenter study describes the features and outcomes of CNO patients in China. The characteristics of CNO children from multiple centers in China were lack of specificity. Bone pain and/or arthralgia were the main clinical symptoms, accompanied by fever, normal white blood cells, and raised inflammatory markers (CRP and/or ESR). Although anti-inflammatory treatment regimens were different, the efficacy of the treatment groups was similar. Anti-inflammatory therapy is currently the first choice.
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spelling doaj.art-05de0893b2274d9bb596ac6b0ad713dd2023-04-06T02:10:58ZzhoEditorial Office of Medical Journal of Peking Union Medical College HospitalXiehe Yixue Zazhi1674-90812023-03-0114227828410.12290/xhyxzz.2022-0712Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in ChinaLIU Haimei0ZHANG Tianyu1MA Le2ZHANG Zhiyong3XU Meng4ZHANG Tao5XU Hong6TANG Xuemei7YANG Sirui8YU Haiguo9SONG Hongmei10SUN Li11National Children's Medical Center, Department of Rheumatology, Children's Hospital of Fudan University, Shanghai 201102, ChinaDepartment of Pediatrics, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, ChinaDepartment of Rheumatology & Immunology, Children's Hospital of Nanjing Medical University, Nanjing 210008, ChinaDepartment of Rheumatology & Immunology, Children's Hospital of Chongqing Medical University, Chongqing 400014, ChinaDepartment of Pediatric Rheumatology, Immunology & Allergy, The First Hospital of Jilin University, Changchun 130021, ChinaNational Children's Medical Center, Department of Rheumatology, Children's Hospital of Fudan University, Shanghai 201102, ChinaNational Children's Medical Center, Department of Rheumatology, Children's Hospital of Fudan University, Shanghai 201102, ChinaDepartment of Rheumatology & Immunology, Children's Hospital of Chongqing Medical University, Chongqing 400014, ChinaDepartment of Pediatric Rheumatology, Immunology & Allergy, The First Hospital of Jilin University, Changchun 130021, ChinaDepartment of Rheumatology & Immunology, Children's Hospital of Nanjing Medical University, Nanjing 210008, ChinaDepartment of Pediatrics, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, ChinaNational Children's Medical Center, Department of Rheumatology, Children's Hospital of Fudan University, Shanghai 201102, ChinaObjective  To investigate the clinical features and treatment outcomes of chronic non-bacterial osteomyelitis (CNO) from five tertiary pediatric rheumatology services in China and provide possible treatment options for clinicians.  Methods  In this multicenter, retrospective study, pediatric patients diagnosed with CNO in Children's Hospital of Fudan University, Peking Union Medical College Hospital, Children's Hospital of Nanjing Medical University, Children's Hospital of Chongqing Medical University and the First Hospital of Jilin University from March 2014 to August 2022 were included. According to the treatment plan, the patients were divided into nonsteroidal anti-inflammatory drugs (NSAID) group, traditional disease modifying anti-rheumatic drugs (DMARD) group, tumor necrosis factor inhibitor (TNF-i) group, bisphosphonate group, and bisphosphonate+TNF-i group, and physician global assessment score was used to evaluate the efficacy.  Results  Retrospective data analysis showed that the disease was more common in boy sthan girls(31 vs. 27), and the average age at onset of symptoms was 8.5±3.4 years, the mean age of diagnosis was 9.8±3.2 years, and the Median follow-up was 10.9(5.0, 30.1) months. A total of 46 cases (79.3%) were diagnosed in the last 3 years. Bone pain and / or arthralgia was the predominant symptom in 54(93.1%) patients followed by fever in 31(53.4%) patients. Raised inflammatory markers (ESR and CRP) were present in 87.9% and 77.6% of the patients. The most frequently affected bones were lower limb bones, including 42 cases of femur and 41 cases of tibia; 3 patients had compression fractures of the vertebrae at the time of diagnosis. Bone biopsy was conducted in 33 cases, and subacute or chronic osteomyelitis manifested with inflammatory cells infiltration were detected. The remission rate was not statistically different between treatment groups (P=0.562), with the remission rate at 11 months in the bisphosphonate+TNF-i group being 60.0%, 55.6% and 33.3% in the NSAID and DMARD groups respectively; 66.7% and 52.4% in the NSAID and TNF-i groups respectively at 12 months. A total of 40 patients were followed up for ≥6 months, with the remission rate of 81.3%.  Conclusions  Our multicenter study describes the features and outcomes of CNO patients in China. The characteristics of CNO children from multiple centers in China were lack of specificity. Bone pain and/or arthralgia were the main clinical symptoms, accompanied by fever, normal white blood cells, and raised inflammatory markers (CRP and/or ESR). Although anti-inflammatory treatment regimens were different, the efficacy of the treatment groups was similar. Anti-inflammatory therapy is currently the first choice.https://xhyxzz.pumch.cn/en/article/doi/10.12290/xhyxzz.2022-0712childrenchronic nonbacterial osteomyelitisclinical featurestreatment outcomes
spellingShingle LIU Haimei
ZHANG Tianyu
MA Le
ZHANG Zhiyong
XU Meng
ZHANG Tao
XU Hong
TANG Xuemei
YANG Sirui
YU Haiguo
SONG Hongmei
SUN Li
Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
Xiehe Yixue Zazhi
children
chronic nonbacterial osteomyelitis
clinical features
treatment outcomes
title Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
title_full Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
title_fullStr Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
title_full_unstemmed Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
title_short Clinical Features and Treatment Outcomes of Chronic Nonbacterial Osteomyelitis in Children: A Multicenter Study in China
title_sort clinical features and treatment outcomes of chronic nonbacterial osteomyelitis in children a multicenter study in china
topic children
chronic nonbacterial osteomyelitis
clinical features
treatment outcomes
url https://xhyxzz.pumch.cn/en/article/doi/10.12290/xhyxzz.2022-0712
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