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成骨不全症年轻女性的上颌骨骨髓炎:揭示一种不常见的关联。

Maxillary osteomyelitis in a young female with osteopetrosis: unveiling an uncommon connection.

机构信息

Department of Dentistry, All India Institute Of Medical Sciences, Mangalagiri, Andhra Pradesh, India

Department of Dentistry, All India Institute Of Medical Sciences, Mangalagiri, Andhra Pradesh, India.

出版信息

BMJ Case Rep. 2023 Oct 31;16(10):e257908. doi: 10.1136/bcr-2023-257908.

Abstract

Osteopetrosis encompasses a spectrum of conditions marked by heightened bone density due to faulty osteoclast-mediated bone resorption, leading to an accumulation of immature bone and thickened cortical structures. This condition gives rise to bone fragility, blood cell irregularities, nerve entrapment and growth challenges, all stemming from disrupted bone remodelling. Craniofacial distinctiveness, encompassing anomalies in the skull and jaw, is a frequent occurrence. Osteopetrosis presents a range of clinical signs, including facial and dental anomalies. The diagnostic process involves thorough clinical and radiological assessments, often obviating the need for genetic testing. Interestingly, few prior reports have delved into the specifics of craniofacial and dental issues in osteopetrosis. The presented case showcases rare occurrence of maxillary osteomyelitis. The diagnosis was established through a combination of history, clinical, radiographic and laboratory findings. The patient declined surgical intervention, leading to the implementation of conservative management involving regular irrigation alongside systemic antibiotic therapy.

摘要

石骨症是一组疾病,其特征为破骨细胞介导的骨吸收功能缺陷导致骨密度增高,从而导致未成熟骨的积累和皮质结构增厚。这种情况导致骨脆性增加、血细胞异常、神经受压和生长挑战,所有这些都源于骨重塑的破坏。颅面特征,包括颅骨和颌骨的异常,是常见的。石骨症表现出一系列临床体征,包括面部和牙齿异常。诊断过程包括全面的临床和影像学评估,通常不需要进行基因检测。有趣的是,很少有先前的报告深入研究石骨症的颅面和牙齿问题。本病例展示了上颌骨骨髓炎的罕见发生。通过病史、临床、影像学和实验室检查结果的综合分析,确立了诊断。患者拒绝手术干预,因此采取了保守治疗,包括定期冲洗和全身抗生素治疗。

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本文引用的文献

1
Osteopetrosis with Arnold Chiari malformation type I.成骨不全伴 Arnold-Chiari 畸形 I 型。
BMJ Case Rep. 2023 Aug 16;16(8):e254559. doi: 10.1136/bcr-2023-254559.
10
Maxillary osteomyelitis associated with osteopetrosis: Systematic review.上颌骨骨髓炎伴骨质硬化症:系统评价。
J Craniomaxillofac Surg. 2018 Nov;46(11):1905-1910. doi: 10.1016/j.jcms.2018.09.004. Epub 2018 Sep 24.

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