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股骨骨齿发育不良中非骨化性纤维瘤的严重表现。

Severe presentation of non-ossifying fibroma of the femur in osteoglophonic dysplasia.

机构信息

Orthopaedics, Hospital Universiti Kebangsaan Malaysia, Kuala Lumpur, Malaysia.

Orthopaedics, Universiti Putra Malaysia Fakulti Perubatan dan Sains Kesihatan, Serdang, Selangor, Malaysia.

出版信息

BMJ Case Rep. 2021 Nov 5;14(11):e245415. doi: 10.1136/bcr-2021-245415.

Abstract

Osteoglophonic dwarfism, also known as osteoglophonic dysplasia (OD), is an uncommon skeletal dysplasia with an autosomal dominant mode of inheritance, which equally affects boys and girls. OD is saliently featured by craniosynostosis, dysmorphic facial features, impacted mandibular teeth, rhizomelic limb shortening and non-ossifying fibromas habitually at the metaphyseal regions, which usually disappear after skeletal maturity. The long bones in OD are portrayed by this distinguishable 'hollowed-out' appearance with metaphyseal cystic defects that have a natural history of spontaneous resolution. We report a case of a rare and unusual presentation of OD in a 23-year-old woman whom has been diagnosed with OD during her early childhood. She presented with a progressively enlarging right thigh swelling associated with pain for the past 1 year. Her right femur plain radiograph revealed diffuse lysis of the whole femur with cortical thinning. MRI revealed multiple bilateral femur benign cystic lesion synonymous with a severe spectrum of OD. She was started on a trial of oral bisphosphonates, which led to a significant improvement in pain.

摘要

骨齿发育不全症,又称骨齿发育不良(OD),是一种罕见的常染色体显性遗传骨骼发育不良疾病,男女发病率均等。OD 的特征为颅缝早闭、面部畸形、下颌牙齿阻生、四肢骨干短小、干骺端非骨化性纤维瘤,这些特征通常在骨骼成熟后消失。OD 的长骨表现为“中空”外观,干骺端囊性缺损,具有自发缓解的自然病程。我们报告了一例罕见且不寻常的骨齿发育不全症病例,患者为一名 23 岁女性,在幼儿时期被诊断为骨齿发育不全症。她因过去 1 年来右大腿逐渐增大的肿胀伴疼痛就诊。她的右股骨正位片显示整个股骨弥漫性溶解,皮质变薄。MRI 显示双侧股骨多发良性囊性病变,与严重的骨齿发育不良症谱一致。她开始口服双膦酸盐治疗,疼痛显著改善。

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本文引用的文献

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Osteoglophonic Dysplasia: Phenotypic and Radiological Clues.骨舌发育不良:表型和影像学线索
J Pediatr Genet. 2017 Dec;6(4):247-251. doi: 10.1055/s-0037-1602816. Epub 2017 May 5.
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Osteoglophonic dysplasia: A 'common' mutation in a rare disease.骨颌发育不良:一种罕见疾病中的“常见”突变。
Clin Genet. 2010 Aug;78(2):197-8. doi: 10.1111/j.1399-0004.2010.01382.x. Epub 2010 Mar 5.
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Bisphosphonate therapy in fibrous dysplasia.纤维性骨营养不良中的双膦酸盐治疗
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