| Literature DB >> 25652355 |
Misako Kunii1, Hiroshi Doi1, Yuichi Higashiyama1, Chiharu Kugimoto1, Naohisa Ueda1, Junichi Hirata1, Atsuko Tomita-Katsumoto1, Mari Kashikura-Kojima1, Shun Kubota1, Midori Taniguchi2, Kei Murayama2, Mitsuko Nakashima3, Yoshinori Tsurusaki3, Noriko Miyake3, Hirotomo Saitsu3, Naomichi Matsumoto3, Fumiaki Tanaka1.
Abstract
Mitochondrial complex III (CIII) deficiency comprises a group of complex and heterogeneous genetic disorders. TTC19 mutations constitute a rare cause of CIII deficiency and are associated with neurological disorders in childhood and adulthood. Herein, we describe a 27-year-old Japanese man with cerebellar ataxia, spastic paraparesis, loss of deep sensation, mild frontal lobe dysfunction and transient psychiatric symptoms. Brain magnetic resonance imaging showed cerebellar atrophy and bilateral high-intensity signals in the inferior olives and regions adjacent to periaqueductal gray matter, on T2-weighted images. On whole-exome sequencing, we detected a novel homozygous frameshift mutation c.157_158dup [p.Pro54Alafs*48] in TTC19. Mitochondrial enzyme assays confirmed mild impairment of CIII enzymatic activity in lymphoblasts, which was consistent with TTC19-related CIII deficiency. His symptoms and radiological findings demonstrated an early stage or mild form of this disease, and further clarify the characteristics of patients with rare TTC19 mutations.Entities:
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Year: 2015 PMID: 25652355 DOI: 10.1038/jhg.2015.7
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172