Literature DB >> 9195224

Nine novel L1 CAM mutations in families with X-linked hydrocephalus.

J R MacFarlane1, J S Du, M E Pepys, S Ramsden, D Donnai, R Charlton, C Garrett, J Tolmie, J R Yates, C Berry, D Goudie, A Moncla, P Lunt, S Hodgson, M Jouet, S Kenwrick.   

Abstract

Mutations in the gene for neural cell adhesion molecule L1 are responsible for the highly variable phenotype found in families with X-linked hydrocephalus, MASA syndrome, and spastic paraplegia type I. To date, 32 different mutations have been observed, the majority being unique to individual families. Here, we report nine novel mutations in L1 in 10 X-linked hydrocephalus families. Four mutations truncate the L1 protein and eliminate cell surface expression, and two would produce abnormal L1 through alteration of RNA processing. A further two of these mutations are small in-frame deletions that have occurred through a mechanism involving tandem repeated sequences. Together with a single missense mutation, these latter examples contribute to the growing number of existing mutations that affect short regions of the L1 protein that may have particular functional significance.

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Year:  1997        PMID: 9195224     DOI: 10.1002/(SICI)1098-1004(1997)9:6<512::AID-HUMU3>3.0.CO;2-3

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  3 in total

1.  The site of a missense mutation in the extracellular Ig or FN domains of L1CAM influences infant mortality and the severity of X linked hydrocephalus.

Authors:  R C Michaelis; Y Z Du; C E Schwartz
Journal:  J Med Genet       Date:  1998-11       Impact factor: 6.318

2.  Detection of L1 CAM mutation in a male child with mental retardation.

Authors:  M Swarna; M Sujatha; P Usha Rani; P P Reddy
Journal:  Indian J Clin Biochem       Date:  2004-07

3.  Severe Phenotype in Patients with X-linked Hydrocephalus Caused by a Missense Mutation in L1CAM.

Authors:  Beyhan Tüysüz; Adife Gülhan Ercan-Sençicek; Emre Özer; Nükte Göç; Cengiz Yalçınkaya; Kaya Bilguvar
Journal:  Turk Arch Pediatr       Date:  2022-09
  3 in total

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