TY - JOUR
T1 - Measuring saccades in patients with Niemann-Pick type C
T2 - A comparison between video-oculography and a novel eye tracking test based on continuous psychophysics
AU - Grillini, A.
AU - Koens, L. H.
AU - Lizaitiene, G.
AU - Lange, F.
AU - Cornelissen, F. W.
AU - Tijssen, M. A.J.
N1 - Funding Information:
We thank the Niemann-Pick type C patients and healthy controls for participating in this study. M.A.J.T is a member of the European Reference Network for Rare Neurological Diseases - Project ID No 739510.”, LHK and FL report no disclosures. AG is the majority shareholder of REPERIO B.V. a private company that develops ophthalmic and neurological tests based on eye movements. AG is listed as inventor on the European patent application Grillini, A. Hernández-García, A. Renken, J. R. (2019). Method, system and computer program product for mapping a visual field. EP19209204.7 which is partially based on the content of this manuscript. GL was supported by a Research Experience Fellowship for 2020 from the European Academy of Neurology. FWC reports grants from the European Union's Horizon 2020 research and innovation programme (Marie Sklodowska-Curie(grant agreement No 641805 “NextGenVis”)), NOVUM, Programmaraad Visuele Sector, Uitzicht (collaborating funds), Graduate School Medical Sciences (GSMS), University of Groningen. MAJT reports grants from the Netherlands Organisation for Health Research and Development ZonMW Topsubsidie (91218013), the European Fund for Regional Development from the European Union (01492947), and the province of Friesland, Dystonia Medical Research Foundation, from Stichting Wetenschapsfonds Dystonie Vereniging, from Fonds Psychische Gezondheid, from Phelps Stichting, and an unrestricted grants from Actelion and AOP Orphan Pharmaceuticals AG. The authors confirm that the approval of an institutional review board was obtained (METc 2018/593 and METc 2016/16). Every participant signed informed consent. AG is the majority shareholder of REPERIO B.V. a private company that develops diagnostic tests for Glaucoma based on eye movements. AG is listed as inventor on the European patent application Grillini, A. Hernández-García, A. Renken, J. R. (2019). Method, system, and computer program product for mapping a visual field. EP19209204.7 which is partially related to the technique presented in this manuscript.
Funding Information:
FWC reports grants from the European Union’s Horizon 2020 research and innovation programme (Marie Sklodowska-Curie(grant agreement No 641805 “NextGenVis”)), NOVUM, Programmaraad Visuele Sector, Uitzicht (collaborating funds), Graduate School Medical Sciences (GSMS), University of Groningen.
Funding Information:
GL was supported by a Research Experience Fellowship for 2020 from the European Academy of Neurology.
Funding Information:
MAJT reports grants from the Netherlands Organisation for Health Research and Development ZonMW Topsubsidie (91218013), the European Fund for Regional Development from the European Union (01492947), and the province of Friesland, Dystonia Medical Research Foundation, from Stichting Wetenschapsfonds Dystonie Vereniging, from Fonds Psychische Gezondheid, from Phelps Stichting, and an unrestricted grants from Actelion and AOP Orphan Pharmaceuticals AG.
Publisher Copyright:
© 2022
PY - 2022/1
Y1 - 2022/1
N2 - Introduction: Vertical supranuclear gaze palsy is a key feature of Niemann-Pick type C (NP-C) and is commonly quantified using video-oculography (VOG). VOG requires sitting still for long times and performing specific tasks, thus it can be challenging or impossible for patients severely affected by movement disorders or cognitive impairment. To overcome this limitation, we measure saccades of NP-C patients using a fast eye tracking test based on continuous psychophysics and compare it to VOG. Methods: Saccades of six NP-C patients and six age-matched controls were assessed using VOG and Standardized Oculomotor and Neuro-ophthalmic Disorders Assessment (SONDA). In SONDA, participants continuously track a semi-randomly moving dot on a computer screen while their gaze is being tracked. For both assessments, saccades were quantified using four conventional measures: amplitude, gain, latency, and peak velocity. Furthermore, SONDA's continuous measures were quantified with several novel spatio-temporal properties. Results: In the NP-C patients, both methods revealed reduced amplitude, gain, peak velocity, and increased latency of vertical saccades compared to horizontal saccades and compared to healthy controls. Effect sizes obtained with SONDA were overall larger than those for VOG. SONDA's spatio-temporal properties showed similar trends. Conclusion: SONDA reveals a deterioration of vertical saccades in NP-C patients that is consistent with VOG. SONDA's measures based on continuous psychophysics are consistent with traditional saccadic parameters and can potentially provide complementary information. SONDA shows larger effect sizes than VOG, suggesting that it provides robust and clinically relevant outcomes with a more intuitive task and shorter testing time.
AB - Introduction: Vertical supranuclear gaze palsy is a key feature of Niemann-Pick type C (NP-C) and is commonly quantified using video-oculography (VOG). VOG requires sitting still for long times and performing specific tasks, thus it can be challenging or impossible for patients severely affected by movement disorders or cognitive impairment. To overcome this limitation, we measure saccades of NP-C patients using a fast eye tracking test based on continuous psychophysics and compare it to VOG. Methods: Saccades of six NP-C patients and six age-matched controls were assessed using VOG and Standardized Oculomotor and Neuro-ophthalmic Disorders Assessment (SONDA). In SONDA, participants continuously track a semi-randomly moving dot on a computer screen while their gaze is being tracked. For both assessments, saccades were quantified using four conventional measures: amplitude, gain, latency, and peak velocity. Furthermore, SONDA's continuous measures were quantified with several novel spatio-temporal properties. Results: In the NP-C patients, both methods revealed reduced amplitude, gain, peak velocity, and increased latency of vertical saccades compared to horizontal saccades and compared to healthy controls. Effect sizes obtained with SONDA were overall larger than those for VOG. SONDA's spatio-temporal properties showed similar trends. Conclusion: SONDA reveals a deterioration of vertical saccades in NP-C patients that is consistent with VOG. SONDA's measures based on continuous psychophysics are consistent with traditional saccadic parameters and can potentially provide complementary information. SONDA shows larger effect sizes than VOG, suggesting that it provides robust and clinically relevant outcomes with a more intuitive task and shorter testing time.
KW - Eye movement disorders
KW - Niemann-Pick type C
KW - SONDA
KW - Vertical supranuclear gaze palsy
KW - VOG
U2 - 10.1016/j.prdoa.2022.100170
DO - 10.1016/j.prdoa.2022.100170
M3 - Article
AN - SCOPUS:85141845396
SN - 2590-1125
VL - 7
JO - Clinical Parkinsonism and Related Disorders
JF - Clinical Parkinsonism and Related Disorders
M1 - 100170
ER -