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手部慢性复发性多灶性骨髓炎:一种罕见的儿科疾病。

Chronic Recurrent Multifocal Osteomyelitis of the Hand: A Rare Pediatric Condition.

作者信息

Karunaratne Yasiru G, Davies Jonathan, Carty Christopher P, Graham David

机构信息

St Vincent's Hospital, Sydney, NSW, Australia.

Queensland Children's Hospital, Brisbane, Australia.

出版信息

Hand (N Y). 2021 Mar;16(2):213-222. doi: 10.1177/1558944719846599. Epub 2019 May 29.

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory condition characterized by bone pain and swelling, secondary to sterile bone osteolytic lesions. Estimated incidence is 1:1 000 000, with ~2% involving the hand. We present a case series of CRMO of the hand and review the available literature, with the aim of educating and raising awareness of this condition. A retrospective chart review was conducted for all patients presenting to the institution diagnosed with CRMO involving the hands. Subsequently, a literature review was performed. Three cases of CRMO of the hand were identified. Age ranged from 20 months to 6 years, with 1 female. Time from presentation to diagnosis ranged from 3 to 15 months. Two had a single lesion, with the remaining patient having a second lesion on full-body magnetic resonance imaging (MRI). Three lesions involved phalanges, and one involved a metacarpal. Two received antibiotics without improvement prior to diagnosis. After diagnosis of CRMO, two received intravenous pamidronate. They both later showed improvement on MRI. CRMO is a rare disease with scarce literature to guide management. We present a series of CRMO specifically of the hand. The cases educate key stakeholders, raise awareness of the diagnosis, and illustrate challenging aspects of managing these patients. This includes the special functional and anatomical consideration essential in managing cases involving the hand.

摘要

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,其特征为骨痛和肿胀,继发于无菌性骨溶解性病变。估计发病率为1:1 000 000,约2%累及手部。我们报告一组手部CRMO病例系列并回顾现有文献,旨在对这种疾病进行教育并提高认识。对本机构诊断为手部CRMO的所有患者进行了回顾性病历审查。随后进行了文献综述。确定了3例手部CRMO病例。年龄范围为20个月至6岁,其中1例为女性。从就诊到诊断的时间为3至15个月。2例有单个病灶,其余患者在全身磁共振成像(MRI)检查时发现有第二个病灶。3个病灶累及指骨,1个累及掌骨。2例在诊断前接受抗生素治疗但无改善。诊断为CRMO后,2例接受了静脉注射帕米膦酸二钠治疗。他们后来在MRI上均显示有改善。CRMO是一种罕见疾病,指导治疗的文献很少。我们报告了一系列专门针对手部的CRMO病例。这些病例对关键利益相关者进行了教育,提高了对该诊断的认识,并说明了管理这些患者的挑战性方面。这包括在处理涉及手部的病例时必不可少的特殊功能和解剖学考虑因素。

相似文献

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Chronic recurrent multifocal osteomyelitis: A rare entity.慢性复发性多灶性骨髓炎:一种罕见病症。
J Med Imaging Radiat Oncol. 2015 Aug;59(4):436-444. doi: 10.1111/1754-9485.12311. Epub 2015 Apr 21.

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