Literature DB >> 30800049

COQ8A and MED25 Mutations in a Child with Intellectual Disability, Microcephaly, Seizures, and Spastic Ataxia: Synergistic Effect of Digenic Variants?

Pratibha Nair1, Maher Lama2, Stephany El-Hayek1, Gretta Abou Sleymane3, Samantha Stora4, Marc Obeid3, Mahmoud T Al-Ali1, Valérie Delague5, André Mégarbané4.   

Abstract

We report on a girl, born to first-cousin Lebanese parents, with severe intellectual disability, congenital hip luxation, cardiac malformation, short stature, facial dysmorphic features including microcephaly, sparse hair, bilateral epicanthal folds, ataxia, seizures, and elevated lactate and pyruvate levels in serum. Whole exome sequencing was carried out on the patient's DNA. Potentially causal homozygous variants in the MED25 (p.Ile173Thr) and COQ8A (p.Arg512Trp) genes were found. The potential pathogenicity of these variants, and the possibility that the 2 variants could synergistically act to produce the phenotype reported, is discussed.

Entities:  

Keywords:  Cerebellar ataxia; Dysmorphology; Whole exome sequencing

Year:  2018        PMID: 30800049      PMCID: PMC6381915          DOI: 10.1159/000494465

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  2 in total

1.  Improving the phenotype description of Basel-Vanagaite-Smirin-Yosef syndrome, MED25-related: polymicrogyria as a distinctive neuroradiological finding.

Authors:  Ilenia Maini; Edoardo Errichiello; Stefano Giuseppe Caraffi; Orsetta Zuffardi; Livia Garavelli; Simonetta Rosato; Veronica Bizzarri; Marzia Pollazzon; Gabriele Trimarchi; Gianluca Contrò; Benedetta Cavirani; Chiara Gelmini; Manuela Napoli; Claudio Moratti; Rosario Pascarella; Susanna Rizzi; Carlo Fusco
Journal:  Neurogenetics       Date:  2020-08-20       Impact factor: 2.660

2.  Report of a Second Lebanese Family with Basel-Vanagaite-Smirin-Yosef Syndrome: Possible Founder Mutation.

Authors:  Pratibha Nair; Sandra Sabbagh; Sami Bizzari; Florian Brunner; Samantha Stora; Mahmoud T Al-Ali; Martin Gencik; Stephany El-Hayek; André Mégarbané
Journal:  Mol Syndromol       Date:  2019-06-28
  2 in total

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