A new variant in the NALCN channel is responsible for cerebellar ataxia and cognitive impairment

CLIFAHDD syndrome (OMIM # 616266) is a rare neurodevelopmental disorder caused by mutations in the NALCN gene. It is characterized by hypotonia, developmental delay, and congenital contractures of the limbs and face. We report a 33-year-old Italian woman with a mild form of CLIFAHDD who exhibited early-onset language difficulties and mild intellectual disability and later developed gait and balance impairments in adulthood. Whole Exome Sequencing (WES) identified a novel missense variant c.1514A>T; p.(Lys505Met) in the NALCN gene. The allele frequency of this variant is not detected (MAF = 0.0), the variant is classified as likely pathogenic according to ACMG criteria, and predicted to be probably damaging by PolyPhen-2. It affects a critical residue within the second pore-forming domain of the NALCN channel, potentially altering lipid interactions and channel regulation. Sanger sequencing and segregation analysis confirmed the variant to be heterozygous and de novo. The patient's milder symptoms and later onset, compared to severe pediatric cases, suggest that the clinical spectrum of CLIFAHDD syndrome may be broader than previously recognized. These findings underscore the potential influence of mutation location on disease presentation and severity.

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Publication Details

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Portuguese National Funding Agency for Science, Research and Technology (RCAAP Project by FCT)
Published
2025-10-11
Primary Topic
Mitochondrial Function and Pathology
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article
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article

A new variant in the NALCN channel is responsible for cerebellar ataxia and cognitive impairment

Angela Elvira Covone, Silvia Viaggi, Giuseppina Conteduca, Barbara Testa et al.
Portuguese National Funding Agency for Science, Research and Technology (RCAAP Project by FCT)
Mitochondrial Function and Pathology
article

A new variant in the NALCN channel is responsible for cerebellar ataxia and cognitive impairment

Angela Elvira Covone, Silvia Viaggi, Giuseppina Conteduca, Barbara Testa, Roberto Fancellu, Tiziana Benzi Markushi, Rute Luísa Cabrita Pinto, Domenico Coviello, Lane Fitzsimmons
article en

Abstract

CLIFAHDD syndrome (OMIM # 616266) is a rare neurodevelopmental disorder caused by mutations in the NALCN gene. It is characterized by hypotonia, developmental delay, and congenital contractures of the limbs and face. We report a 33-year-old Italian woman with a mild form of CLIFAHDD who exhibited early-onset language difficulties and mild intellectual disability and later developed gait and balance impairments in adulthood. Whole Exome Sequencing (WES) identified a novel missense variant c.1514A>T; p.(Lys505Met) in the NALCN gene. The allele frequency of this variant is not detected (MAF = 0.0), the variant is classified as likely pathogenic according to ACMG criteria, and predicted to be probably damaging by PolyPhen-2. It affects a critical residue within the second pore-forming domain of the NALCN channel, potentially altering lipid interactions and channel regulation. Sanger sequencing and segregation analysis confirmed the variant to be heterozygous and de novo. The patient's milder symptoms and later onset, compared to severe pediatric cases, suggest that the clinical spectrum of CLIFAHDD syndrome may be broader than previously recognized. These findings underscore the potential influence of mutation location on disease presentation and severity.

Portuguese National Funding Agency for Science, Research and Technology (RCAAP Project by FCT)
Quality Education
Openalex Percentile: Top 35%
Mitochondrial Function and Pathology
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A new variant in the NALCN channel is responsible for cerebellar ataxia and cognitive impairment — Angela Elvira Covone, Silvia Viaggi, et al. · Portuguese National Funding Agency for Science, Research and Technology (RCAAP Project by FCT) (2025) | TGRS Research Map | TGRS