| Literature DB >> 23633388 |
Leonie Speksnijder1, Titia E Cohen-Overbeek, Maarten F C M Knapen, Simone M Lunshof, A Jeannette M Hoogeboom, Ans M van den Ouwenland, Irenaneus F M de Coo, Maarten H Lequin, Hanno J Bolz, Carsten Bergmann, Leslie G Biesecker, Patrick J Willems, Marja W Wessels.
Abstract
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.Entities:
Mesh:
Substances:
Year: 2013 PMID: 23633388 DOI: 10.1002/ajmg.a.35874
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802