变态发育不良的广泛临床谱:一个病例系列和文献回顾。

IF 4.9 2区 生物学
Kiabeth Robles-Espinoza, Eduardo Esparza-García, Juan Ramón González García, María Teresa Magaña-Torres
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引用次数: 0

摘要

萎缩性发育不良是一种常染色体显性骨骼疾病,其特征为进行性脊柱后凸、严重的平椎、明显的干骺端增大和长骨缩短。这种情况是由TRPV4(瞬时受体电位香草蛋白4)基因的致病性变异引起的,该基因编码参与骨稳态的非选择性钙通道。TRPV4的变异与两种主要疾病组相关:骨骼发育不良和神经病变,最近的研究结果表明它们的临床特征有重叠。我们报告了三例异萎缩性发育不良患者,每个患者都表现出不同的严重程度。所有人都有钟形胸,明显的平椎,短而长的骨,骨外宽。值得注意的是,患者1和3有更复杂的临床过程,包括癫痫发作和整体发育迟缓。遗传分析显示两种不同的TRPV4变体:p.Asn796del(患者1)和p.Pro799Leu(患者2和3)。这些病例说明了骨骼外表现、并发症和预后的可变性。在我们的trpv4相关疾病患者中,神经系统症状和骨骼异常的共同出现表明临床异质性谱与单一疾病一致,而不是不同的实体。综合的、多学科的方法对于优化管理和提高患者的生活质量至关重要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

The Broad Clinical Spectrum of Metatropic Dysplasia: A Case Series and Literature Review.

The Broad Clinical Spectrum of Metatropic Dysplasia: A Case Series and Literature Review.

The Broad Clinical Spectrum of Metatropic Dysplasia: A Case Series and Literature Review.

Metatropic dysplasia is an autosomal dominant skeletal disorder characterized by progressive kyphoscoliosis, severe platyspondyly, pronounced metaphyseal enlargement, and shortening of the long bones. This condition is caused by pathogenic variants in the TRPV4 (Transient Receptor Potential Vanilloid 4) gene, which encodes a non-selective calcium channel involved in bone homeostasis. Variants in TRPV4 have been associated with two major disease groups: skeletal dysplasias and neuropathies, with recent findings indicating an overlap in their clinical features. We report three patients with metatropic dysplasia, each presenting a distinct severity profile. All exhibited a bell-shaped thorax, significant platyspondyly, and shortened long bones with broad metaphyses. Notably, patients 1 and 3 had more complex clinical courses, including seizures and global developmental delay. Genetic analysis revealed two different TRPV4 variants: p.Asn796del (patient 1) and p.Pro799Leu (patients 2 and 3). These cases illustrate variability in extra-skeletal manifestations, complications, and prognosis. In our patients with TRPV4-related disorders, the co-occurrence of neurological symptoms and skeletal abnormalities suggests a clinically heterogeneous spectrum consistent with a single disease rather than distinct entities. A comprehensive, multidisciplinary approach is essential to optimize management and improve the quality of life for patients.

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来源期刊
自引率
10.70%
发文量
13472
审稿时长
1.7 months
期刊介绍: The International Journal of Molecular Sciences (ISSN 1422-0067) provides an advanced forum for chemistry, molecular physics (chemical physics and physical chemistry) and molecular biology. It publishes research articles, reviews, communications and short notes. Our aim is to encourage scientists to publish their theoretical and experimental results in as much detail as possible. Therefore, there is no restriction on the length of the papers or the number of electronics supplementary files. For articles with computational results, the full experimental details must be provided so that the results can be reproduced. Electronic files regarding the full details of the calculation and experimental procedure, if unable to be published in a normal way, can be deposited as supplementary material (including animated pictures, videos, interactive Excel sheets, software executables and others).
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