Literature DB >> 9215766

Evidence for genetic anticipation in the oculodentodigital syndrome.

R E Shapiro1, J W Griffin, O C Stine.   

Abstract

Oculodentodigital syndrome (O.D.) is an autosomal dominant disorder comprising facial anomalies, syndactyly, microcorneae, dental enamel hypoplasia, and leukodystrophy. We describe a four generation family with O.D. in which anomalies such as syndactyly appear congenitally, whereas neurological (i.e., leukodystrophic) signs and symptoms tend to be expressed in a more severe form and/or at an earlier age of onset in successive generations of the kindred. This pattern of phenotypic expression is consistent with the phenomenon of genetic anticipation, and we suggest that O.D. may be a trinucleotide repeat disorder.

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Mesh:

Year:  1997        PMID: 9215766

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  9 in total

Review 1.  Gap junctions couple astrocytes and oligodendrocytes.

Authors:  Jennifer L Orthmann-Murphy; Charles K Abrams; Steven S Scherer
Journal:  J Mol Neurosci       Date:  2008-05       Impact factor: 3.444

2.  A novel truncation mutation in GJA1 associated with open angle glaucoma and microcornea in a large Chinese family.

Authors:  X Huang; N Wang; X Xiao; S Li; Q Zhang
Journal:  Eye (Lond)       Date:  2015-05-15       Impact factor: 3.775

Review 3.  Gap junctions in inherited human disorders of the central nervous system.

Authors:  Charles K Abrams; Steven S Scherer
Journal:  Biochim Biophys Acta       Date:  2011-08-16

4.  Oculodentodigital Dysplasia: A Hypomyelinating Leukodystrophy with a Characteristic MRI Pattern of Brain Stem Involvement.

Authors:  I Harting; S Karch; U Moog; A Seitz; P J W Pouwels; N I Wolf
Journal:  AJNR Am J Neuroradiol       Date:  2019-05-02       Impact factor: 3.825

5.  Connexin 43 (GJA1) mutations cause the pleiotropic phenotype of oculodentodigital dysplasia.

Authors:  William A Paznekas; Simeon A Boyadjiev; Robert E Shapiro; Otto Daniels; Bernd Wollnik; Catherine E Keegan; Jeffrey W Innis; Mary Beth Dinulos; Cathy Christian; Mark C Hannibal; Ethylin Wang Jabs
Journal:  Am J Hum Genet       Date:  2002-11-27       Impact factor: 11.025

Review 6.  Variable expression of neurological phenotype in autosomal recessive oculodentodigital dysplasia of two sibs and review of the literature.

Authors:  Shelagh K Joss; Sam Ghazawy; Susan Tomkins; Mushtaq Ahmed; John Bradbury; Eamonn Sheridan
Journal:  Eur J Pediatr       Date:  2007-05-03       Impact factor: 3.183

7.  Heterozygous GJA1 variants with ocular phenotype: Missense in domain but truncation out of domain.

Authors:  Xueqing Li; Xueshan Xiao; Shiqiang Li; Jiamin Ouyang; Wenmin Sun; Xing Liu; Qingjiong Zhang
Journal:  Mol Vis       Date:  2021-05-13       Impact factor: 2.367

8.  Oculodentodigital Dysplasia: A Case Report and Major Review of the Eye and Ocular Adnexa Features of 295 Reported Cases.

Authors:  Virang Kumar; Natario L Couser; Arti Pandya
Journal:  Case Rep Ophthalmol Med       Date:  2020-04-04

9.  Specific functional pathologies of Cx43 mutations associated with oculodentodigital dysplasia.

Authors:  John J Kelly; Jessica L Esseltine; Qing Shao; Ethylin Wang Jabs; Jacinda Sampson; Mari Auranen; Donglin Bai; Dale W Laird
Journal:  Mol Biol Cell       Date:  2016-05-25       Impact factor: 4.138

  9 in total

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