| Literature DB >> 25454649 |
Amir Jahic1, Friedmar Kreuz2, Pia Zacher3, Jana Fiedler2, Andrea Bier2, Silke Reif2, Manuela Rieger2, Stefan Krüger2, Christian Beetz4, Jens Plaschke2.
Abstract
Hereditary spastic paraplegia (HSP) is a clinically and genetically heterogeneous, neurodegenerative movement disorder. A total of eight KIAA0196/strumpellin variants have thus far been associated with SPG8, a rare dominant HSP. We present a novel strumpellin alteration in a small family with clinically pure HSP. We corroborated its causality by comparing it to rare benign variants at several levels, and, along this line, also re-considered previous genetic reports on SPG8. These analyses identified significant challenges in the interpretation of strumpellin alterations, and suggested that at least two of the few families claimed to suffer from SPG8 may have been genetically misdiagnosed.Entities:
Keywords: Hereditary spastic paraplegic; KIAA0196; Pathogenicity prediction; SPG8; Strumpellin
Mesh:
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Year: 2014 PMID: 25454649 DOI: 10.1016/j.jns.2014.10.018
Source DB: PubMed Journal: J Neurol Sci ISSN: 0022-510X Impact factor: 3.181