Literature DB >> 29737427

Serum neurofilament light is increased in multiple system atrophy of cerebellar type and in repeat-expansion spinocerebellar ataxias: a pilot study.

Carlo Wilke1,2, Friedemann Bender1,2, Stefanie N Hayer1,2, Kathrin Brockmann1,2, Ludger Schöls1,2, Jens Kuhle3, Matthis Synofzik4,5.   

Abstract

Blood biomarkers in degenerative ataxias are still largely missing. Here, we aimed to provide piloting proof-of-concept that serum Neurofilament light (NfL) could offer a promising peripheral blood biomarker in degenerative ataxias. Specifically, as a marker of neuronal damage, NfL might (1) help to differentiate multiple system atrophy of cerebellar type (MSA-C) from sporadic adult-onset ataxia (SAOA), and (2) show increases in repeat-expansion spinocerebellar ataxias (SCAs) which might be amenable to treatment in the future. To explore these two hypotheses, we measured serum NfL levels by single-molecule array (Simoa) technique in 115 subjects, comprising patients with MSA-C (n = 25), SAOA (n = 25), the most frequent repeat-expansion SCAs (SCA 1, 2, 3 and 6) (n = 20), and age-matched controls (n = 45). Compared to controls, NfL was significantly increased in MSA-C, with levels significantly higher than in SAOA (AUC = 0.74 (0.59-0.89), mean and 95% confidence interval, p = .004). NfL was also significantly increased in SCA patients as compared to controls (AUC = 0.91 (0.81-1.00), p < .001), including NfL increases in SCA1 and SCA3. These findings provide first proof-of-concept that NfL might provide a promising peripheral biomarker in degenerative ataxias, e.g. supporting the differentiation of MSA-C from SAOA, and indicating neuronal damage in repeat-expansion SCAs.

Entities:  

Keywords:  Biomarker; Multiple system atrophy of cerebellar type (MSA-C); Neurofilament light chain (NfL); Serum; Spinocerebellar ataxia (SCA); Sporadic adult-onset ataxia (SAOA)

Mesh:

Substances:

Year:  2018        PMID: 29737427     DOI: 10.1007/s00415-018-8893-9

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  21 in total

1.  The natural history of spinocerebellar ataxia type 1, 2, 3, and 6: a 2-year follow-up study.

Authors:  H Jacobi; P Bauer; P Giunti; R Labrum; M G Sweeney; P Charles; A Dürr; C Marelli; C Globas; C Linnemann; L Schöls; M Rakowicz; R Rola; E Zdzienicka; T Schmitz-Hübsch; R Fancellu; C Mariotti; C Tomasello; L Baliko; B Melegh; A Filla; C Rinaldi; B P van de Warrenburg; C C P Verstappen; S Szymanski; J Berciano; J Infante; D Timmann; S Boesch; S Hering; C Depondt; M Pandolfo; J-S Kang; S Ratzka; J Schulz; S Tezenas du Montcel; T Klockgether
Journal:  Neurology       Date:  2011-08-10       Impact factor: 9.910

2.  Neurofilament light chain in FTD is elevated not only in cerebrospinal fluid, but also in serum.

Authors:  Carlo Wilke; Oliver Preische; Christian Deuschle; Benjamin Roeben; Anja Apel; Christian Barro; Luis Maia; Walter Maetzler; Jens Kuhle; Matthis Synofzik
Journal:  J Neurol Neurosurg Psychiatry       Date:  2016-05-17       Impact factor: 10.154

3.  CSF neurofilament light differs in neurodegenerative diseases and predicts severity and survival.

Authors:  Tobias Skillbäck; Bahman Farahmand; Jonathan W Bartlett; Christoffer Rosén; Niklas Mattsson; Katarina Nägga; Lena Kilander; Dorota Religa; Anders Wimo; Bengt Winblad; Lars Rosengren; Jonathan M Schott; Kaj Blennow; Maria Eriksdotter; Henrik Zetterberg
Journal:  Neurology       Date:  2014-10-22       Impact factor: 9.910

4.  Neurofilament light chain level in cerebrospinal fluid can differentiate Parkinson's disease from atypical parkinsonism: Evidence from a meta-analysis.

Authors:  Wataru Sako; Nagahisa Murakami; Yuishin Izumi; Ryuji Kaji
Journal:  J Neurol Sci       Date:  2015-04-07       Impact factor: 3.181

5.  Clinical and genetic characteristics of sporadic adult-onset degenerative ataxia.

Authors:  Ilaria Giordano; Florian Harmuth; Heike Jacobi; Brigitte Paap; Stefan Vielhaber; Judith Machts; Ludger Schöls; Matthis Synofzik; Marc Sturm; Chantal Tallaksen; Iselin M Wedding; Sylvia Boesch; Andreas Eigentler; Bart van de Warrenburg; Judith van Gaalen; Christoph Kamm; Ales Dudesek; Jun-Suk Kang; Dagmar Timmann; Gabriella Silvestri; Marcella Masciullo; Thomas Klopstock; Christiane Neuhofer; Christos Ganos; Alessandro Filla; Peter Bauer; Sophie Tezenas du Montcel; Thomas Klockgether
Journal:  Neurology       Date:  2017-08-09       Impact factor: 9.910

6.  Neurofilament Light Chain in Blood and CSF as Marker of Disease Progression in Mouse Models and in Neurodegenerative Diseases.

Authors:  Mehtap Bacioglu; Luis F Maia; Oliver Preische; Juliane Schelle; Anja Apel; Stephan A Kaeser; Manuel Schweighauser; Timo Eninger; Marius Lambert; Andrea Pilotto; Derya R Shimshek; Ulf Neumann; Philipp J Kahle; Matthias Staufenbiel; Manuela Neumann; Walter Maetzler; Jens Kuhle; Mathias Jucker
Journal:  Neuron       Date:  2016-06-09       Impact factor: 17.173

7.  Second consensus statement on the diagnosis of multiple system atrophy.

Authors:  S Gilman; G K Wenning; P A Low; D J Brooks; C J Mathias; J Q Trojanowski; N W Wood; C Colosimo; A Dürr; C J Fowler; H Kaufmann; T Klockgether; A Lees; W Poewe; N Quinn; T Revesz; D Robertson; P Sandroni; K Seppi; M Vidailhet
Journal:  Neurology       Date:  2008-08-26       Impact factor: 9.910

8.  Antisense Oligonucleotide-Mediated Removal of the Polyglutamine Repeat in Spinocerebellar Ataxia Type 3 Mice.

Authors:  Lodewijk J A Toonen; Frank Rigo; Haico van Attikum; Willeke M C van Roon-Mom
Journal:  Mol Ther Nucleic Acids       Date:  2017-06-29       Impact factor: 8.886

9.  Antisense oligonucleotide therapy for spinocerebellar ataxia type 2.

Authors:  Daniel R Scoles; Pratap Meera; Matthew D Schneider; Sharan Paul; Warunee Dansithong; Karla P Figueroa; Gene Hung; Frank Rigo; C Frank Bennett; Thomas S Otis; Stefan M Pulst
Journal:  Nature       Date:  2017-04-12       Impact factor: 49.962

10.  Serum Neurofilament light: A biomarker of neuronal damage in multiple sclerosis.

Authors:  Giulio Disanto; Christian Barro; Pascal Benkert; Yvonne Naegelin; Sabine Schädelin; Antonella Giardiello; Chiara Zecca; Kaj Blennow; Henrik Zetterberg; David Leppert; Ludwig Kappos; Claudio Gobbi; Jens Kuhle
Journal:  Ann Neurol       Date:  2017-06       Impact factor: 10.422

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  20 in total

Review 1.  Diagnostic utility of fluid biomarkers in multiple system atrophy: a systematic review and meta-analysis.

Authors:  Shengri Cong; Chunchen Xiang; Hailong Wang; Shuyan Cong
Journal:  J Neurol       Date:  2020-03-11       Impact factor: 4.849

2.  Brain proteome profiling implicates the complement and coagulation cascade in multiple system atrophy brain pathology.

Authors:  Rasmus Rydbirk; Ole Østergaard; Jesper V Olsen; Susana Aznar; Jonas Folke; Casper Hempel; Brian DellaValle; Thomas L Andresen; Annemette Løkkegaard; Anne-Mette Hejl; Matthias Bode; Morten Blaabjerg; Mette Møller; Erik H Danielsen; Lisette Salvesen; Charlotte C Starhof; Sara Bech; Kristian Winge; Jørgen Rungby; Bente Pakkenberg; Tomasz Brudek
Journal:  Cell Mol Life Sci       Date:  2022-06-03       Impact factor: 9.207

3.  Levels of Neurofilament Light at the Preataxic and Ataxic Stages of Spinocerebellar Ataxia Type 1.

Authors:  Carlo Wilke; David Mengel; Ludger Schöls; Holger Hengel; Maria Rakowicz; Thomas Klockgether; Alexandra Durr; Alessandro Filla; Bela Melegh; Rebecca Schüle; Kathrin Reetz; Heike Jacobi; Matthis Synofzik
Journal:  Neurology       Date:  2022-03-09       Impact factor: 11.800

4.  Toward allele-specific targeting therapy and pharmacodynamic marker for spinocerebellar ataxia type 3.

Authors:  Mercedes Prudencio; Hector Garcia-Moreno; Karen R Jansen-West; Rana Hanna Al-Shaikh; Tania F Gendron; Michael G Heckman; Matthew R Spiegel; Yari Carlomagno; Lillian M Daughrity; Yuping Song; Judith A Dunmore; Natalie Byron; Björn Oskarsson; Katharine A Nicholson; Nathan P Staff; Sorina Gorcenco; Andreas Puschmann; João Lemos; Cristina Januário; Mark S LeDoux; Joseph H Friedman; James Polke; Robin Labrum; Vikram Shakkottai; Hayley S McLoughlin; Henry L Paulson; Takuya Konno; Osamu Onodera; Takeshi Ikeuchi; Mari Tada; Akiyoshi Kakita; John D Fryer; Christin Karremo; Inês Gomes; John N Caviness; Mark R Pittelkow; Jan Aasly; Ronald F Pfeiffer; Venka Veerappan; Eric R Eggenberger; William D Freeman; Josephine F Huang; Ryan J Uitti; Klaas J Wierenga; Iris V Marin Collazo; Philip W Tipton; Jay A van Gerpen; Marka van Blitterswijk; Guojun Bu; Zbigniew K Wszolek; Paola Giunti; Leonard Petrucelli
Journal:  Sci Transl Med       Date:  2020-10-21       Impact factor: 17.956

Review 5.  Rating scales and biomarkers for CAG-repeat spinocerebellar ataxias: Implications for therapy development.

Authors:  Meng-Ling Chen; Chih-Chun Lin; Liana S Rosenthal; Puneet Opal; Sheng-Han Kuo
Journal:  J Neurol Sci       Date:  2021-04-01       Impact factor: 3.181

Review 6.  Spinocerebellar ataxia clinical trials: opportunities and challenges.

Authors:  Sarah M Brooker; Chandrakanth Reddy Edamakanti; Sara M Akasha; Sheng-Han Kuo; Puneet Opal
Journal:  Ann Clin Transl Neurol       Date:  2021-05-21       Impact factor: 4.511

Review 7.  Recent advances in understanding dominant spinocerebellar ataxias from clinical and genetic points of view.

Authors:  Giulia Coarelli; Alexis Brice; Alexandra Durr
Journal:  F1000Res       Date:  2018-11-12

8.  DRPLA: understanding the natural history and developing biomarkers to accelerate therapeutic trials in a globally rare repeat expansion disorder.

Authors:  Aiysha Chaudhry; Alkyoni Anthanasiou-Fragkouli; Henry Houlden
Journal:  J Neurol       Date:  2020-10-26       Impact factor: 4.849

9.  Serum neurofilament light chain as a severity marker for spinocerebellar ataxia.

Authors:  Hye-Rim Shin; Jangsup Moon; Woo-Jin Lee; Han Sang Lee; Eun Young Kim; Seoyi Shin; Soon-Tae Lee; Keun-Hwa Jung; Kyung-Il Park; Ki-Young Jung; Sang Kun Lee; Kon Chu
Journal:  Sci Rep       Date:  2021-06-29       Impact factor: 4.379

10.  Neurofilaments in spinocerebellar ataxia type 3: blood biomarkers at the preataxic and ataxic stage in humans and mice.

Authors:  Christian Barro; Jeannette Hübener-Schmid; Matthis Synofzik; Carlo Wilke; Eva Haas; Kathrin Reetz; Jennifer Faber; Hector Garcia-Moreno; Magda M Santana; Bart van de Warrenburg; Holger Hengel; Manuela Lima; Alessandro Filla; Alexandra Durr; Bela Melegh; Marcella Masciullo; Jon Infante; Paola Giunti; Manuela Neumann; Jeroen de Vries; Luis Pereira de Almeida; Maria Rakowicz; Heike Jacobi; Rebecca Schüle; Stephan A Kaeser; Jens Kuhle; Thomas Klockgether; Ludger Schöls
Journal:  EMBO Mol Med       Date:  2020-06-08       Impact factor: 12.137

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