| Literature DB >> 16272165 |
K E Chandler1, A Del Rio, K Rakshi, K Springell, D K Williams, N Stoodley, C G Woods, D T Pilz.
Abstract
We report three related and one unrelated child with an apparently novel neurodevelopmental disorder. The clinical course was very similar in all the four patients: congenital microcephaly with severe failure of post-natal brain growth, neonatal onset of intractable seizures associated with lack of developmental progression and death within the first 3 years of life. The appearance on cerebral neuroimaging was almost identical, with simplified gyration associated with a non-thickened cortex, severe hypoplasia of the corpus callosum, a small flattened brain stem, and specific cystic lesions in the white matter around the temporal and occipital horns. To our knowledge these patients represent a previously unreported, autosomal recessive syndrome. Homozygosity mapping in the consanguineous family has identified a candidate region on the chromosome 2p16.Entities:
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Year: 2005 PMID: 16272165 DOI: 10.1093/brain/awh663
Source DB: PubMed Journal: Brain ISSN: 0006-8950 Impact factor: 13.501