| Literature DB >> 23430795 |
Adela Della Marina1, Ulrike Schara, Angela Pyle, Claudia Möller-Hartmann, Elke Holinski-Feder, Angela Abicht, Birgit Czermin, Hanns Lochmüller, Helen Griffin, Mauro Santibanez-Koref, Patrick F Chinnery, Rita Horvath.
Abstract
With over 1,000 nuclear genes that could potentially cause a mitochondrial disorder, the current diagnostic approach requires targeted molecular analysis, guided by a combination of clinical and biochemical features. However, the expanding molecular and clinical spectrum means that this approach does not always yield a result. Here we report the unusual clinical presentation of "Progressive External Ophthalmoplegia (PEO) plus" Leigh syndrome in three children from a consanguineous family where exome sequencing identified mutations in NDUFS8. NDUFS8 is a nuclear-encoded structural core protein of complex I, and mutations are expected to cause infantile onset and severe disease. Our patients had a later onset, milder and a clinically distinct phenotype, and this gene would not normally be considered in this context. Being untargeted to specific genes, whole exome analysis has the potential to re-write the phenotype and reveal an unexpected molecular aetiology, as illustrated by this family.Entities:
Year: 2012 PMID: 23430795 PMCID: PMC3755572 DOI: 10.1007/8904_2012_195
Source DB: PubMed Journal: JIMD Rep ISSN: 2192-8304