Literature DB >> 25454649

A novel strumpellin mutation and potential pitfalls in the molecular diagnosis of hereditary spastic paraplegia type SPG8.

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.
Copyright © 2014 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Hereditary spastic paraplegic; KIAA0196; Pathogenicity prediction; SPG8; Strumpellin

Mesh:

Substances:

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


  7 in total

Review 1.  SPG8 mutations in Italian families: clinical data and literature review.

Authors:  Federica Ginanneschi; Angelica D'Amore; Melissa Barghigiani; Alessandra Tessa; Alessandro Rossi; Filippo Maria Santorelli
Journal:  Neurol Sci       Date:  2019-12-09       Impact factor: 3.307

2.  Three novel mutations in 20 patients with hereditary spastic paraparesis.

Authors:  Mehmet Bugrahan Duz; Selcuk Dasdemir; Aysel Kalayci Yigin; Mehmet Ali Akalin; Mehmet Seven
Journal:  Neurol Sci       Date:  2018-06-16       Impact factor: 3.307

3.  First patient with hereditary spastic paraplegia type 8 in Poland.

Authors:  Piotr Bogucki; Agnieszka Sobczyńska-Tomaszewska
Journal:  Clin Case Rep       Date:  2017-07-25

4.  Expression of N471D strumpellin leads to defects in the endolysosomal system.

Authors:  Lin Song; Ramesh Rijal; Malte Karow; Maria Stumpf; Oliver Hahn; Laura Park; Robert Insall; Rolf Schröder; Andreas Hofmann; Christoph S Clemen; Ludwig Eichinger
Journal:  Dis Model Mech       Date:  2018-09-13       Impact factor: 5.758

5.  Next-generation sequencing study reveals the broader variant spectrum of hereditary spastic paraplegia and related phenotypes.

Authors:  Ewelina Elert-Dobkowska; Iwona Stepniak; Wioletta Krysa; Karolina Ziora-Jakutowicz; Maria Rakowicz; Anna Sobanska; Jacek Pilch; Dorota Antczak-Marach; Jacek Zaremba; Anna Sulek
Journal:  Neurogenetics       Date:  2019-02-19       Impact factor: 2.660

6.  The spectrum of KIAA0196 variants, and characterization of a murine knockout: implications for the mutational mechanism in hereditary spastic paraplegia type SPG8.

Authors:  Amir Jahic; Mukhran Khundadze; Nadine Jaenisch; Rebecca Schüle; Sven Klimpe; Stephan Klebe; Christiane Frahm; Jan Kassubek; Giovanni Stevanin; Ludger Schöls; Alexis Brice; Christian A Hübner; Christian Beetz
Journal:  Orphanet J Rare Dis       Date:  2015-11-16       Impact factor: 4.123

7.  A novel KIAA0196 mutation in a Chinese patient with spastic paraplegia 8: A case report.

Authors:  Limin Ma; Yingying Shi; Zhongcan Chen; Shujian Li; Weiwei Qin; Jiewen Zhang
Journal:  Medicine (Baltimore)       Date:  2018-05       Impact factor: 1.889

  7 in total

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