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常染色体显性遗传性骨硬化症相关双颌骨髓炎的长期随访:一例报告

Long-Term Follow-Up of Bimaxillary Osteomyelitis Associated with Autosomal Dominant Osteopetrosis: A Case Report.

作者信息

Kulyapina A, Verdaguer Martin J, Navarro Cuellar C, Navarro Vila C

机构信息

Department of Maxillofacial Surgery, Gregorio Marañón General University Hospital, C/Doctor Esquerdo 46, 28007 Madrid, Spain.

出版信息

J Maxillofac Oral Surg. 2016 Mar;15(1):121-6. doi: 10.1007/s12663-015-0749-6. Epub 2015 Feb 10.

DOI:10.1007/s12663-015-0749-6
PMID:26929564
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4759028/
Abstract

Osteopetrosis (OP) is a rare metabolic bone disease characterized by a generalized increase in skeletal mass. The disease is characterized by increased susceptibility to develop osteomyelitis of the jaws. We report a case of clinical and radiological progression of bimaxillary osteomyelitis in a patient with autosomal dominant OP. The patient presented non simultaneous osteomyelitis in both upper and lower jaws with time interval of 10 years. The osteomyelitis of maxilla resulted in oroantral fistula formation and required surgical closure with Bichat fat pad flap. The mandibular osteomyelitis resulted in sequestra formation and pathological fracture and required multiple debridement procedures. Both maxillary and mandibular osteomyelitic foci were persistent and healing required more than 2 years. The case confirms the possibility of development of non simultaneous bimaxillary osteomyelitis in patients with OP. The case enhances the need of close follow- up and preventive measures in patients with OP.

摘要

骨质石化症(OP)是一种罕见的代谢性骨病,其特征是骨骼质量普遍增加。该疾病的特点是易发生颌骨骨髓炎。我们报告一例常染色体显性骨质石化症患者双颌骨髓炎的临床和影像学进展情况。该患者上下颌非同时发生骨髓炎,时间间隔为10年。上颌骨髓炎导致口鼻瘘形成,需要用比沙氏脂肪垫瓣进行手术闭合。下颌骨髓炎导致死骨形成和病理性骨折,需要多次清创手术。上颌和下颌的骨髓炎病灶持续存在,愈合需要超过2年时间。该病例证实了骨质石化症患者发生非同时性双颌骨髓炎的可能性。该病例凸显了对骨质石化症患者进行密切随访和采取预防措施的必要性。

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本文引用的文献

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Osteopetrosis.骨质石化症
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Marginal resection for treatment of mandibular osteomyelitis associated with osteopetrosis: case report.边缘切除术治疗伴有骨质硬化症的下颌骨骨髓炎:病例报告。
J Craniomaxillofac Surg. 2011 Oct;39(7):525-9. doi: 10.1016/j.jcms.2010.10.015. Epub 2010 Nov 11.
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Genetics of bone diseases: Paget's disease, fibrous dysplasia, osteopetrosis, and osteogenesis imperfecta.骨病的遗传学:佩吉特病、纤维结构不良、骨硬化症和成骨不全症。
Joint Bone Spine. 2011 May;78(3):252-8. doi: 10.1016/j.jbspin.2010.07.010. Epub 2010 Sep 19.
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Intractable bimaxillary osteomyelitis in osteopetrosis: review of the literature and current therapy.骨质石化症中的难治性双颌骨髓炎:文献综述与当前治疗方法
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9
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