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Heterozygous p.Asp50Asn mutation in the GJB2 gene in two Cameroonian patients with keratitis-ichthyosis-deafness (KID) syndrome

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  • معلومة اضافية
    • بيانات النشر:
      BioMed Central Ltd
      University of Cape Town
      Faculty of Health Sciences
      Division of Human Genetics
    • الموضوع:
      2013
    • Collection:
      University of Cape Town: OpenUCT
    • نبذة مختصرة :
      BACKGROUND: Keratitis-Ichthyosis-Deafness (KID) syndrome (OMIM 148210) is a congenital ectodermal defect that consists of an atypical ichthyosiform erythroderma associated with congenital sensorineural deafness. KID appears to be genetically heterogeneous and most cases are caused by GJB2 mutations. Mutations in African patients have been rarely described.CASE PRESENTATION:We report on two unrelated Cameroonian individuals affected with sporadic KID, presenting with the classic phenotypic triad. The two patients were heterozygous for the most frequent p.Asp50Asn mutation. This first report in patients from sub-Saharan African origin supports the hypothesis that the occurrence of KID due to p.Asp50Asn mutation in GJB2 seems not to be population specific. CONCLUSIONS: Our finding has implication in medical genetic practice, specifically in the molecular diagnosis of KID in Africans. These cases also reveal and emphasize the urgent need to develop appropriate policies to care for patients with rare/orphan diseases in Sub-Saharan Africa, as many of these cases become more and more recognizable.
    • File Description:
      application/pdf
    • Relation:
      http://hdl.handle.net/11427/15387; http://dx.doi.org/10.1186/1471-2350-14-81; https://open.uct.ac.za/bitstream/11427/15387/1/Wonkam_Heterozygous_pAsp50Asn_2013.pdf
    • الرقم المعرف:
      10.1186/1471-2350-14-81
    • الدخول الالكتروني :
      http://hdl.handle.net/11427/15387
      https://doi.org/10.1186/1471-2350-14-81
      https://open.uct.ac.za/bitstream/11427/15387/1/Wonkam_Heterozygous_pAsp50Asn_2013.pdf
    • Rights:
      This is an Open Access article distributed under the terms of the Creative Commons Attribution License ; http://creativecommons.org/licenses/by/2.0 ; 2013 Wonkam et al.; licensee BioMed Central Ltd.
    • الرقم المعرف:
      edsbas.9FC0A381