Koenig Rainer, Bach Alexia, Woelki Ulrike, Grzeschik Karl-Heinz, Fuchs Sigrun
Department of Human Genetics, Johann Wolfgang Goethe University, Frankfurt, Germany.
Am J Med Genet. 2002 Feb 15;108(1):7-11. doi: 10.1002/ajmg.10236.
Acrocallosal syndrome (ACS) is an autosomal recessive condition, characterized by agenesis of the corpus callosum, pre- and postaxial polydactyly, minor craniofacial anomalies, and, in most patients, severe psychomotor retardation. We here report on three patients with ACS demonstrating a spectrum from mild to severe involvement. Two patients had only mild to moderate mental retardation at the age of 2(1/2) and 4 years, respectively, with surprisingly good speech development. The third patient was severely affected and died at age 7 days because of persistent apnea. All three patients had agenesis of the corpus callosum, and large intracranial cysts, which in the third case was confirmed as a large arachnoid cyst at autopsy. Cranial cysts were also seen in 10/34 published cases of ACS. Thus, intracerebral cysts are a common finding in ACS and may serve in differentiating ACS from Greig cephalopolysyndactyly syndrome.
胼胝体发育不全综合征(ACS)是一种常染色体隐性疾病,其特征为胼胝体发育不全、轴前和轴后多指畸形、轻微颅面异常,且大多数患者存在严重精神运动发育迟缓。我们在此报告3例ACS患者,其病情呈现从轻到重的不同程度。两名患者分别在2岁半和4岁时仅有轻度至中度智力发育迟缓,但其语言发育情况出人意料地良好。第三名患者病情严重,因持续性呼吸暂停在7日龄时死亡。所有3例患者均有胼胝体发育不全及较大的颅内囊肿,在第三例中,尸检证实为巨大蛛网膜囊肿。在已发表的34例ACS病例中,有10例也发现了颅内囊肿。因此,脑内囊肿是ACS的常见表现,可用于将ACS与Greig头多指综合征相鉴别。