Speksnijder Leonie, Cohen-Overbeek Titia E, Knapen Maarten F C M, Lunshof Simone M, Hoogeboom A Jeannette M, van den Ouwenland Ans M, de Coo Irenaneus F M, Lequin Maarten H, Bolz Hanno J, Bergmann Carsten, Biesecker Leslie G, Willems Patrick J, Wessels Marja W
Department of Obstetrics and Gynecology, Erasmus MC University Medical Center, Rotterdam, The Netherlands.
Am J Med Genet A. 2013 Jun;161A(6):1394-400. doi: 10.1002/ajmg.a.35874. Epub 2013 Apr 30.
Acrocallosal syndrome is characterized by postaxial polydactyly, macrocephaly, agenesis of the corpus callosum, and severe developmental delay. In a few patients with this disorder, a mutation in the KIF7 gene has been reported, which was associated with impaired GLI3 processing and dysregulaton of GLI3 transcription factors. A single patient with acrocallosal syndrome and a de novo p.Ala934Pro mutation in GLI3 has been reported, whereas diverse and numerous GLI3 mutations have also been described in syndromes with overlapping clinical manifestations, including Greig cephalopolysyndactyly syndrome, Pallister-Hall syndrome, trigonocephaly with craniosynostosis and polydactyly, oral-facial-digital syndrome, and non-syndromic polydactyly. Here, we describe a second patient with acrocallosal syndrome, who has a de novo, novel c.2786T>C mutation in GLI3, which predicts p.Leu929Pro. This mutation is in the same domain as the mutation in the previously reported patient. These data confirm that mutations in GLI3 are a cause of the acrocallosal phenotype.
胼胝体发育不全综合征的特征为轴后多指畸形、巨头畸形、胼胝体发育不全及严重发育迟缓。在少数患有这种疾病的患者中,已报道KIF7基因突变,这与GLI3加工受损及GLI3转录因子失调有关。曾报道过1例患有胼胝体发育不全综合征且GLI3基因发生新生p.Ala934Pro突变的患者,而在具有重叠临床表现的综合征中也描述了多种GLI3突变,包括Greig头面多指综合征、帕利斯特-霍尔综合征、三角头畸形伴颅缝早闭和多指畸形、口面指综合征及非综合征性多指畸形。在此,我们描述了第2例患有胼胝体发育不全综合征的患者,其GLI3基因发生了新生的、新的c.2786T>C突变,预测为p.Leu929Pro。该突变与先前报道患者的突变位于同一结构域。这些数据证实GLI3突变是胼胝体发育不全表型的一个病因。