Mierzewska Hanna, Mierzewska-Schmidt Magdalena, Salomons Gajja S, Dudzińska Magdalena, Szczepanik Elżbieta
Department of Child and Adolescent Neurology, Institute of Mother and Child, Clinic of Child and Adolescent Neurology-IMD Kasprzaka 17A, 01-211 Warsaw, Poland tel/fax: (+48 22) 32-77-130 e-mail:
Dept. of Pediatric Anaesthesiology and Intensive Unit, Warsaw Medical University, Poland.
Dev Period Med. 2016 Apr-Jun;20(2):110-7.
Alexander Disease (ALXDRD) is an autosomal dominant leukodystrophy caused by mutation in one allele of GFAP gene, encoding glial fibrillary acidic protein (GFAP). Most cases occur due to de novo. There are three clinical subtypes of ALXDRD: infantile, juvenile and adult form, but congenital form is also outlined. The disease's spectrum comprises of macrocephaly, progressive pyramidal signs, and seizures in congenital and infantile subtypes. Neuropathologically are enormous number of Rosenthal fibers (RF) mainly around vessels, in subependymal and subpial regions are found. The diagnosis is based on the typical findings on MRI: diffuse white mater lesions with frontal regions preponderance and possibly on the detection of the gene mutation. Here we present six Polish children affected of Alexander disease with congenital (1), infantile (4) and juvenile (1) form. Five of them were previously misdiagnosed as cerebral palsy or unspecific developmental delay; two patients had MRI because of another suspicion, before specific diagnosis was established. Molecular analysis performed in four cases confirmed mutations of GFAP gene; all mutation were de novo. The role of astroglia in brain is shortly reviewed.
亚历山大病(ALXDRD)是一种常染色体显性遗传性脑白质营养不良,由编码胶质纤维酸性蛋白(GFAP)的GFAP基因的一个等位基因突变引起。大多数病例为新发。ALXDRD有三种临床亚型:婴儿型、青少年型和成人型,但也有先天性形式的描述。该疾病的谱系包括大头畸形、先天性和婴儿型亚型中的进行性锥体束征和癫痫发作。神经病理学上,主要在血管周围、室管膜下和软脑膜下区域发现大量的罗森塔尔纤维(RF)。诊断基于MRI上的典型表现:以额叶为主的弥漫性白质病变,以及可能基于基因突变的检测。在此,我们报告6例患有先天性(1例)、婴儿型(4例)和青少年型(1例)亚历山大病的波兰儿童。其中5例之前被误诊为脑瘫或非特异性发育迟缓;在确诊之前,2例患者因其他怀疑进行了MRI检查。对4例患者进行的分子分析证实了GFAP基因的突变;所有突变均为新发。本文简要回顾了星形胶质细胞在大脑中的作用。