Literature DB >> 19725242

Oculodentodigital dysplasia: disease spectrum in an eight-year-old boy, his parents and a sibling.

Naser Asl Aminabadi1, Azin Taghizadeh Ganji, Ali Vafaei, Maryam Pourkazemi, Sina Ghertasi Oskouei.   

Abstract

Oculodentodigital dysplasia is an extremely rare autosomal dominant pleiotropic disorder caused by mutations in the Connexin 43 gene (GJA1). Described here is a previously undiagnosed case of an 8-year-old boy with enamel and dentin hypoplasia and typical faces. In this presentation, many typical clinical and radiographical features of this condition are present. The characteristic features include a typical face, premature loss of primary teeth and odontodysplasia of permanent teeth, clinodactyly, ocular signs, and CNS involvement. To our knowledge, the case that we report here is the first case with mamelon-shaped tip of the tongue and enlarged midpalatal raphe.

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Year:  2009        PMID: 19725242     DOI: 10.17796/jcpd.33.4.0r02810u1533h168

Source DB:  PubMed          Journal:  J Clin Pediatr Dent        ISSN: 1053-4628            Impact factor:   1.065


  2 in total

1.  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

2.  Epilepsy as a rare neurologic manifestation of oculodentodigitalis dysplasia.

Authors:  Mohammad Barzegar; Mohammad Sayadnasiri; Aidin Tabrizi
Journal:  Iran J Child Neurol       Date:  2012
  2 in total

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