de Wit M C Y, de Coo I F M, Halley D J J, Lequin M H, Mancini G M S
Department of Pediatric Neurology, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands.
Neurogenetics. 2009 Oct;10(4):333-6. doi: 10.1007/s10048-009-0192-2. Epub 2009 Apr 22.
We report a child with a severe choreadystonic movement disorder, bilateral periventricular nodular heterotopia (BPNH), and secondary microcephaly based on compound heterozygosity for two new ARFGEF2 mutations (c.2031_2038dup and c.3798_3802del), changing the limited knowledge about the phenotype. The brain MRI shows bilateral hyperintensity of the putamen, BPNH, and generalized atrophy. Loss of ARFGEF2 function affects vesicle trafficking, proliferation/apoptosis, and neurotransmitter receptor function. This can explain BPNH and microcephaly. We hypothesize that the movement disorder and the preferential damage to the basal ganglia, specifically to the putamen, may be caused by an increased sensitivity to degeneration, a dynamic dysfunction due to neurotransmitter receptor mislocalization or a combination of both.
我们报告了一名患有严重舞蹈样肌张力障碍运动障碍、双侧脑室周围结节性异位(BPNH)和继发性小头畸形的儿童,其基于两个新的ARFGEF2突变(c.2031_2038dup和c.3798_3802del)的复合杂合性,改变了对该表型的有限认知。脑部MRI显示壳核双侧高信号、BPNH和广泛性萎缩。ARFGEF2功能丧失会影响囊泡运输、增殖/凋亡和神经递质受体功能。这可以解释BPNH和小头畸形。我们推测,运动障碍以及对基底神经节,特别是壳核的优先损害,可能是由于对退化的敏感性增加、神经递质受体定位错误导致的动态功能障碍或两者共同作用引起的。