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一个家族中存在 TTN 缺失,该家族患有骨骼肌病、面肌无力和扩张型心肌病。

A novel TTN deletion in a family with skeletal myopathy, facial weakness, and dilated cardiomyopathy.

机构信息

The Ohio State University Wexner Medical Center, Columbus, Ohio.

Invitae Corporation, San Francisco, California.

出版信息

Mol Genet Genomic Med. 2019 Nov;7(11):e924. doi: 10.1002/mgg3.924. Epub 2019 Sep 5.

Abstract

BACKGROUND

Pathogenic variants in TTN (OMIM 188840), encoding the largest human protein, are known to cause dilated cardiomyopathy and several forms of skeletal myopathy. The clinical interpretation of TTN variants is challenging, however, due to the frequency of missense changes, variable testing and reporting practices in commercial laboratories, and incomplete understanding of the spectrum of TTN-related disease.

METHODS

We report a heterozygous TTN deletion segregating in a family with an unusual skeletal myopathy phenotype associated with facial weakness, gait abnormality, and dilated cardiomyopathy.

RESULTS

A novel 16.430 kb heterozygous deletion spanning part of the A- and M-bands of TTN was identified in the proband and his symptomatic son, as well as in an additional son whose symptoms were identified on clinical evaluation. The deletion was found to be de novo in the proband.

CONCLUSION

Pathogenic variants in TTN may be an unrecognized cause of skeletal myopathy phenotypes, particularly when accompanied by dilated cardiomyopathy.

摘要

背景

编码人类最大蛋白的 TTN(OMIM 188840)中的致病性变异已知可导致扩张型心肌病和几种形式的骨骼肌病。然而,由于同义突变的频率、商业实验室中可变的检测和报告实践以及对 TTN 相关疾病谱的不完全了解,TTN 变异的临床解释具有挑战性。

方法

我们报告了一个杂合 TTN 缺失在一个家族中的分离,该家族具有不寻常的骨骼肌病表型,伴有面部无力、步态异常和扩张型心肌病。

结果

在先证者及其有症状的儿子以及另外一个儿子中发现了一个新的 16.430kb 杂合缺失,该缺失跨越了 TTN 的 A-和 M-带的一部分,该缺失在先证者中为新发。

结论

TTN 中的致病性变异可能是骨骼肌病表型的一个未被认识的原因,特别是当伴有扩张型心肌病时。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e07f/6825852/d1e2b769ec1a/MGG3-7-e924-g001.jpg

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