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慢性非细菌性骨髓炎患儿自发消退的肿块样软组织受累:病例回顾

Spontaneously regressing mass-like soft tissue involvement in a child with chronic non-bacterial osteomyelitis: case-based review.

作者信息

Koru Lutfiye, Baysal Begumhan, Yıldırım Ayse Nur Toksoz, Dizman Eda Nur, Dursun Hatice Kubra, Balci Merve Ozen, Kucuk Elif, Kaya Feray, Aydin Zelal, Haslak Fatih, Ozturk Kubra

机构信息

Department of Pediatric Rheumatology, Istanbul Medeniyet University, Istanbul, Turkey.

Department of Radiology, Istanbul Medeniyet University, Istanbul, Turkey.

出版信息

Rheumatol Int. 2025 Jan 3;45(1):16. doi: 10.1007/s00296-024-05768-1.

DOI:10.1007/s00296-024-05768-1
PMID:39751896
Abstract

Chronic non-bacterial osteomyelitis (CNO) is an inflammatory bone disease, usually diagnosed in childhood. It is characterized by the presence of multifocal or unifocal osteolytic lesions that can cause bone pain and soft tissue swelling. CNO is known to have soft tissue involvement. However, soft tissue involvement large enough to give the appearance of a mass is rare. This article discusses a case of CNO with a mass-like appearance that involved soft tissue and spontaneously regressed, as well as presents CNO cases with soft tissue involvement and conducts a literature review on this subject. Our investigation revealed that edema and synovitis were the most frequently observed soft tissue involvements in association with CNO. Moreover, we also encountered myositis, a mass-like appearance, neuritis, and polyserositis within the surrounding muscles. Although, it is well known that bone inflammation tends to regress spontaneously in CNO patients which reflects the autoinflammatory nature of the disease, there is no such patient whose soft tissue involvement which has mass like appearance improved spontaneously. Therefore, we aimed to emphasize the clinical progress which can be easily underdiagnosed of CNO patients by the clinicians by presenting unique features of our patient and our detailed literature review.

摘要

慢性非细菌性骨髓炎(CNO)是一种炎症性骨病,通常在儿童期被诊断出来。其特征是存在多灶性或单灶性溶骨性病变,可导致骨痛和软组织肿胀。已知CNO会累及软组织。然而,软组织受累范围大到足以形成肿块的情况很少见。本文讨论了一例表现为肿块样外观且累及软组织并自发消退的CNO病例,同时展示了伴有软组织受累的CNO病例,并对该主题进行了文献综述。我们的调查显示,水肿和滑膜炎是与CNO相关的最常见的软组织受累情况。此外,我们还在周围肌肉中发现了肌炎、肿块样外观、神经炎和多浆膜炎。尽管众所周知,CNO患者的骨炎症往往会自发消退,这反映了该疾病的自身炎症性质,但尚无软组织受累呈现肿块样外观的患者自发好转的情况。因此,我们旨在通过展示我们患者的独特特征和详细的文献综述,强调临床医生可能容易漏诊的CNO患者的临床进展情况。

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Spontaneously regressing mass-like soft tissue involvement in a child with chronic non-bacterial osteomyelitis: case-based review.慢性非细菌性骨髓炎患儿自发消退的肿块样软组织受累:病例回顾
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本文引用的文献

1
Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis.慢性非细菌性骨髓炎的临床特征和复发预测因素:一项回顾性多中心分析。
Turk J Med Sci. 2023 Feb 28;53(5):1105-1111. doi: 10.55730/1300-0144.5675. eCollection 2023.
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Chronic recurrent multifocal osteomyelitis of clavicle: a rare isolated location (a case report).锁骨慢性复发性多灶性骨髓炎:罕见的孤立部位(病例报告)。
Pan Afr Med J. 2023 Oct 12;46:53. doi: 10.11604/pamj.2023.46.53.39452. eCollection 2023.
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Classification and management strategies for paediatric chronic nonbacterial osteomyelitis and chronic recurrent multifocal osteomyelitis.
儿童慢性非细菌性骨髓炎和慢性复发性多灶性骨髓炎的分类及管理策略
Expert Rev Clin Immunol. 2023 Jul-Dec;19(9):1101-1116. doi: 10.1080/1744666X.2023.2218088. Epub 2023 Jun 7.
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Neurosurgical management of vertebral lesions in pediatric chronic recurrent multifocal osteomyelitis: patient series.小儿慢性复发性多灶性骨髓炎椎体病变的神经外科治疗:病例系列
J Neurosurg Case Lessons. 2023 Jan 23;5(4). doi: 10.3171/CASE22179.
5
Early diagnosis and response assessment in chronic recurrent multifocal osteomyelitis: changes in lesion volume and signal intensity assessed by whole-body MRI.慢性复发性多灶性骨髓炎的早期诊断和反应评估:全身 MRI 评估病变体积和信号强度的变化。
Br J Radiol. 2022 Feb 1;95(1130):20211091. doi: 10.1259/bjr.20211091. Epub 2021 Dec 16.
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Chronic recurrent multifocal osteomyelitis: A multidisciplinary experience of 22 pediatric cases with a mean follow-up of 27 months.慢性复发性多灶性骨髓炎:22 例儿科病例的多学科经验,平均随访 27 个月。
J Orthop Sci. 2023 Mar;28(2):438-445. doi: 10.1016/j.jos.2021.11.009. Epub 2021 Dec 8.
7
Chronic recurrent multifocal osteomyelitis causing optic neuropathy.慢性复发性多灶性骨髓炎导致视神经病变。
J AAPOS. 2022 Feb;26(1):43-46. doi: 10.1016/j.jaapos.2021.09.003. Epub 2021 Nov 20.
8
Mind the Localized Skeletal Pain: Chronic Recurrent Multifocal Osteomyelitis.注意局部骨骼疼痛:慢性复发性多灶性骨髓炎。
Cureus. 2021 May 18;13(5):e15101. doi: 10.7759/cureus.15101.
9
Primary squamous cell carcinoma of the urinary bladder presenting as an inguinal mass.表现为腹股沟肿块的原发性膀胱鳞状细胞癌。
SA J Radiol. 2021 Mar 29;25(1):2048. doi: 10.4102/sajr.v25i1.2048. eCollection 2021.
10
Chronic nonbacterial osteomyelitis - clinical and magnetic resonance imaging features.慢性非细菌性骨髓炎 - 临床和磁共振成像特征。
Pediatr Radiol. 2021 Feb;51(2):282-288. doi: 10.1007/s00247-020-04827-6. Epub 2020 Oct 9.