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通过临床外显子组测序在一名XI型成骨不全症儿科患者中鉴定出FKBP10的新型突变。

Novel mutation of FKBP10 in a pediatric patient with osteogenesis imperfecta type XI identified by clinical exome sequencing.

作者信息

Velasco Harvy Mauricio, Morales Jessica L

机构信息

Master of Human Genetics, National University of Colombia and Geneticist in Central Military Hospital, Bogotá DC, Colombia.

Faculty of Medicine, Universidad Nacional de Colombia, Bogotá DC, Colombia.

出版信息

Appl Clin Genet. 2017 Nov 7;10:75-83. doi: 10.2147/TACG.S126277. eCollection 2017.

Abstract

Osteogenesis imperfecta (OI) is a hereditary disease characterized by bone fragility caused by mutations in the proteins that support the formation of the extracellular matrix in the bone. The diagnosis of OI begins with clinical suspicion, from phenotypic findings at birth, low-impact fractures during childhood or family history that may lead to it. However, the variability in the semiology of the disease does not allow establishing an early diagnosis in all cases, and unfortunately, specific clinical data provided by the literature only report 28 patients with OI type XI. This information is limited and heterogeneous, and therefore, detailed information on the natural history of this disease is not yet available. This paper reports the case of a male patient who, despite undergoing multidisciplinary management, did not have a diagnosis for a long period of time, and could only be given one with the use of whole-exome sequencing. The use of the next-generation sequencing in patients with ultrarare genetic diseases, including skeletal dysplasias, should be justified when clear clinical criteria and an improvement in the quality of life of the patients and their families are intended while reducing economic and time costs. Thus, this case report corresponds to the 29th patient affected with OI type XI, and the 18th mutation in FKBP10, causative of this pathology.

摘要

成骨不全症(OI)是一种遗传性疾病,其特征是由于支持骨细胞外基质形成的蛋白质发生突变而导致骨脆性增加。OI的诊断始于临床怀疑,这源于出生时的表型表现、儿童期的低冲击力骨折或可能导致该病的家族病史。然而,该疾病症状学的变异性使得无法在所有病例中进行早期诊断,不幸的是,文献中提供的具体临床数据仅报告了28例XI型OI患者。这些信息有限且参差不齐,因此,关于该疾病自然史的详细信息尚不可得。本文报告了一例男性患者的病例,该患者尽管接受了多学科管理,但长期未得到诊断,仅通过全外显子测序才得以确诊。对于患有超罕见遗传病(包括骨骼发育不良)的患者,当旨在明确临床标准并改善患者及其家庭的生活质量,同时降低经济和时间成本时,使用下一代测序技术是合理的。因此,本病例报告对应的是第29例XI型OI患者,也是导致该病理状况的FKBP10基因的第18次突变。

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