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儿童成骨不全症诊断时骨折的临床特征及类型

CLINICAL FEATURES AND PATTERN OF FRACTURES AT THE TIME OF DIAGNOSIS OF OSTEOGENESIS IMPERFECTA IN CHILDREN.

作者信息

Brizola Evelise, Zambrano Marina Bauer, Pinheiro Bruna de Souza, Vanz Ana Paula, Félix Têmis Maria

机构信息

Universidade Federal do Rio Grande do Sul, Porto Alegre, RS, Brasil.

Hospital de Clínicas de Porto Alegre, Porto Alegre, RS, Brasil.

出版信息

Rev Paul Pediatr. 2017 Apr-Jun;35(2):171-177. doi: 10.1590/1984-0462/;2017;35;2;00001.

Abstract

OBJECTIVE

To characterize the fracture pattern and the clinical history at the time of diagnosis of osteogenesis imperfecta.

METHODS

In this retrospective study, all patients with osteogenesis imperfecta, of both genders, aged 0-18 years, who were treated between 2002 and 2014 were included. Medical records were assessed to collect clinical data, including the presence of blue sclerae, dentinogenesis imperfecta, positive familial history of osteogenesis imperfecta, and the site of the fractures. In addition, radiographic findings at the time of the diagnosis were reviewed.

RESULTS

Seventy-six patients (42 females) were included in the study. Individuals' age ranged from 0 to 114 months, with a median (interquartile range) age of 38 (6-96) months. Blue sclerae were present in 93.4% of patients, dentinogenesis imperfecta was observed in 27.6% of patients, and wormian bones in 29.4% of them. The number of fractures at diagnosis ranged from 0 to 17, with a median of 3 (2-8) fractures. Forty (57%) patients had fractures of the upper and lower extremities, and 9 patients also had spinal fractures. The diagnosis was performed at birth in 85.7% of patients with type 3, and 39.3% of those with type 4/5 of the disorder.

CONCLUSIONS

Osteogenesis imperfecta is a genetic disorder with distinctive clinical features such as bone fragility, recurrent fractures, blue sclerae, and dentinogenesis imperfecta. It is important to know how to identify these characteristics in order to facilitate the diagnosis, optimize the treatment, and differentiate osteogenesis imperfecta from other disorders that also can lead to fractures.

摘要

目的

描述成骨不全症诊断时的骨折类型和临床病史。

方法

在这项回顾性研究中,纳入了2002年至2014年间接受治疗的所有0至18岁的成骨不全症患者,不分性别。评估病历以收集临床数据,包括蓝色巩膜、牙本质发育不全、成骨不全症家族史阳性以及骨折部位。此外,还回顾了诊断时的影像学检查结果。

结果

76例患者(42例女性)纳入研究。患者年龄从0至114个月不等,中位(四分位间距)年龄为38(6 - 96)个月。93.4%的患者有蓝色巩膜,27.6%的患者观察到牙本质发育不全,29.4%的患者有缝间骨。诊断时骨折数量从0至17例不等,中位骨折数为3(2 - 8)例。40例(57%)患者有上肢和下肢骨折,9例患者还有脊柱骨折。3型患者中85.7%在出生时确诊,4/5型患者中39.3%在出生时确诊。

结论

成骨不全症是一种遗传性疾病,具有骨脆性、反复骨折、蓝色巩膜和牙本质发育不全等独特临床特征。了解如何识别这些特征对于促进诊断、优化治疗以及将成骨不全症与其他也可导致骨折的疾病相鉴别很重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a0fc/5496728/4b869861ea72/0103-0582-rpp-35-02-00171-gf1.jpg

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