KIF1A-associated neurological disorders: therapeutic opportunities and challenges

KIF1A-Associated Neurological Disorder (KAND) is a rare, progressive neurodegenerative condition caused by variants in the KIF1A gene, which encodes a kinesin-3 motor protein essential for anterograde axonal transport of synaptic vesicles, dense core vesicles, and organelles in neurons. KAND comprises a broad spectrum of overlapping neurological phenotypes, including hereditary spastic paraplegia, intellectual disability, peripheral neuropathy, optic nerve atrophy, epilepsy, and progressive motor decline. Pathogenic variants in KIF1A disrupt the balance of intracellular transport and neuronal signalling through diverse mechanisms, manifesting with highly variable disease onset, severity, and clinical progression. Although advances in genomic testing have improved diagnosis, reported KAND cases remain concentrated in developed countries, highlighting ongoing global inequities in access to diagnosis and care. At present, no cure exists for KAND; treatment is limited to symptom management. A deeper understanding of KIF1A function, supported by the development of robust cellular and animal models, is critical for therapeutic development. This review summarises the clinical and molecular features of KAND and highlights current and emerging strategies aimed at slowing disease progression or correcting its underlying causes. We emphasise the urgent need for targeted treatment strategies addressing the heterogeneity of KAND.

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Publication Details

Journal
European Journal of Human Genetics
Published
2025-11-27
DOI
https://doi.org/10.1038/s41431-025-01978-8
Primary Topic
Hereditary Neurological Disorders
Type
article
Field-Weighted Citation Impact
0.00

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article

KIF1A-associated neurological disorders: therapeutic opportunities and challenges

Wendy Gold, John Christodoulou, Simranpreet Kaur, Qing Lin et al.
European Journal of Human Genetics
Hereditary Neurological Disorders
article

KIF1A-associated neurological disorders: therapeutic opportunities and challenges

Wendy Gold, John Christodoulou, Simranpreet Kaur, Qing Lin, Dylan Verden
article en

Abstract

KIF1A-Associated Neurological Disorder (KAND) is a rare, progressive neurodegenerative condition caused by variants in the KIF1A gene, which encodes a kinesin-3 motor protein essential for anterograde axonal transport of synaptic vesicles, dense core vesicles, and organelles in neurons. KAND comprises a broad spectrum of overlapping neurological phenotypes, including hereditary spastic paraplegia, intellectual disability, peripheral neuropathy, optic nerve atrophy, epilepsy, and progressive motor decline. Pathogenic variants in KIF1A disrupt the balance of intracellular transport and neuronal signalling through diverse mechanisms, manifesting with highly variable disease onset, severity, and clinical progression. Although advances in genomic testing have improved diagnosis, reported KAND cases remain concentrated in developed countries, highlighting ongoing global inequities in access to diagnosis and care. At present, no cure exists for KAND; treatment is limited to symptom management. A deeper understanding of KIF1A function, supported by the development of robust cellular and animal models, is critical for therapeutic development. This review summarises the clinical and molecular features of KAND and highlights current and emerging strategies aimed at slowing disease progression or correcting its underlying causes. We emphasise the urgent need for targeted treatment strategies addressing the heterogeneity of KAND.

European Journal of Human Genetics
The University of Sydney (AU), Royal Children's Hospital (AU), Children's Medical Research Institute (AU), The University of Melbourne (AU), Hubei Institute of Fine Arts (CN), Children's Hospital at Westmead (AU), Victorian Clinical Genetics Services (AU), Sydney Children’s Hospitals Network (AU), Murdoch Children's Research Institute (AU), Neuroscience Research Australia (AU)
Children’s Hospital of Wisconsin Research Institute, Children's Hospital Foundation, Murdoch Children's Research Institute, Royal Children's Hospital Foundation, Australian Government, State Government of Victoria, National Health and Medical Research Council
Openalex Percentile: Top 60%
Hereditary Neurological Disorders
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