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14 号染色体缺失、环状染色体和癫痫基因:一个谜中套着谜的谜。

Chromosome 14 deletions, rings, and epilepsy genes: A riddle wrapped in a mystery inside an enigma.

机构信息

Institute of Genomic Medicine, Catholic University School of Medicine, Rome, Italy.

Residential Center for Rehabilitation Luce Sul Mare, Rimini, Italy.

出版信息

Epilepsia. 2021 Jan;62(1):25-40. doi: 10.1111/epi.16754. Epub 2020 Nov 17.

DOI:10.1111/epi.16754
PMID:33205446
Abstract

The ring 14 syndrome is a rare condition caused by the rearrangement of one chromosome 14 into a ring-like structure. The formation of the ring requires two breakpoints and loss of material from the short and long arms of the chromosome. Like many other chromosome syndromes, it is characterized by multiple congenital anomalies and developmental delays. Typical of the condition are retinal anomalies and drug-resistant epilepsy. These latter manifestations are not found in individuals who are carriers of comparable 14q deletions without formation of a ring (linear deletions). To find an explanation for this apparent discrepancy and gain insight into the mechanisms leading to seizures, we reviewed and compared literature cases of both ring and linear deletion syndrome with respect to both their clinical manifestations and the role and function of potentially epileptogenic genes. Knowledge of the epilepsy-related genes in chromosome 14 is an important premise for the search of new and effective drugs to combat seizures. Current clinical and molecular evidence is not sufficient to explain the known discrepancies between ring and linear deletions.

摘要

14 号环状染色体综合征是一种罕见的疾病,由一条 14 号染色体发生重排形成环状结构引起。形成环状结构需要两个断裂点和染色体短臂和长臂的物质丢失。像许多其他染色体综合征一样,它的特征是多种先天性异常和发育迟缓。该疾病的典型表现是视网膜异常和耐药性癫痫。在没有形成环状结构(线性缺失)的情况下,携带类似的 14q 缺失的个体不会出现这些表现。为了解释这种明显的差异,并深入了解导致癫痫发作的机制,我们回顾和比较了环状和线性缺失综合征的文献病例,比较了它们的临床表现以及潜在致痫基因的作用和功能。了解 14 号染色体上与癫痫相关的基因是寻找新的有效药物来对抗癫痫发作的重要前提。目前的临床和分子证据还不足以解释环状和线性缺失之间已知的差异。

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