Literature DB >> 16272165

Leucodysplasia, microcephaly, cerebral malformation (LMC): a novel recessive disorder linked to 2p16.

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:  

Mesh:

Substances:

Year:  2005        PMID: 16272165     DOI: 10.1093/brain/awh663

Source DB:  PubMed          Journal:  Brain        ISSN: 0006-8950            Impact factor:   13.501


  5 in total

1.  Clinical and molecular cytogenetic characterisation of a newly recognised microdeletion syndrome involving 2p15-16.1.

Authors:  E Rajcan-Separovic; C Harvard; X Liu; B McGillivray; J G Hall; Y Qiao; J Hurlburt; J Hildebrand; E C R Mickelson; J J A Holden; M E S Lewis
Journal:  J Med Genet       Date:  2006-09-08       Impact factor: 6.318

2.  Further characterization of microdeletion syndrome involving 2p15-p16.1.

Authors:  Têmis Maria Félix; Aline Lourenço Petrin; Maria Teresa Vieira Sanseverino; Jeffrey C Murray
Journal:  Am J Med Genet A       Date:  2010-10       Impact factor: 2.802

Review 3.  A developmental and genetic classification for midbrain-hindbrain malformations.

Authors:  A James Barkovich; Kathleen J Millen; William B Dobyns
Journal:  Brain       Date:  2009-12       Impact factor: 13.501

4.  KinSNP software for homozygosity mapping of disease genes using SNP microarrays.

Authors:  El-Ad David Amir; Ofer Bartal; Efrat Morad; Tal Nagar; Jony Sheynin; Ruti Parvari; Vered Chalifa-Caspi
Journal:  Hum Genomics       Date:  2010-08       Impact factor: 4.639

5.  Posterior fossa in primary microcephaly: relationships between forebrain and mid-hindbrain size in 110 patients.

Authors:  Yuko Adachi; Ganeshwaran Mochida; Christopher Walsh; James Barkovich
Journal:  Neuropediatrics       Date:  2013-11-14       Impact factor: 1.947

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.