Analysis of genetic risk factors for Leber hereditary optic neuropathy in the Polish population

Leber hereditary optic neuropathy (LHON) is primarily caused by pathogenic mitochondrial DNA (mtDNA) variants, most commonly the m.11778G>A variant in the MT-ND4 gene. The presence of this variant alone is insufficient to trigger disease symptoms, of which vision loss is the hallmark. Given the incomplete penetrance and inter-population variability in modifying factors, this study aimed to investigate two previously proposed genetic risk factors for LHON in the Polish population. Using quantitative PCR, we measured the mtDNA copy number in peripheral blood of affected and unaffected carriers of the m.11778G>A variant. In addition, we assessed the frequency of the PRICKLE3 c.157C>T variant in symptomatic, asymptomatic and control individuals using PCR-RFLP. Our results indicate that mtDNA copy number was not associated with LHON symptom manifestation in the Polish cohort under conditions tested, in contrast to previously reported associations in other populations. In addition, the PRICKLE3 c.157C>T variant was absent in our cohort, indicating that it is not a common modifier of LHON penetrance in the Polish population. These findings suggest that the incomplete penetrance of LHON in the Polish population may involve other modifying factors, such as yet unidentified nuclear DNA variants.

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Publication Details

Journal
Journal of Applied Genetics
Published
2026-06-27
DOI
https://doi.org/10.1007/s13353-026-01090-7
Primary Topic
Mitochondrial Function and Pathology
Type
article
Field-Weighted Citation Impact
0.00

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article

Analysis of genetic risk factors for Leber hereditary optic neuropathy in the Polish population

Monika Ołdak, Ewa Bartnik, Agnieszka Piotrowska-Nowak, M Krawczyński et al.
Journal of Applied Genetics
Mitochondrial Function and Pathology
article

Analysis of genetic risk factors for Leber hereditary optic neuropathy in the Polish population

Monika Ołdak, Ewa Bartnik, Agnieszka Piotrowska-Nowak, M Krawczyński, Katarzyna Tońska, Magdalena Korwin, Julia A. Sikorska
article en

Abstract

Leber hereditary optic neuropathy (LHON) is primarily caused by pathogenic mitochondrial DNA (mtDNA) variants, most commonly the m.11778G>A variant in the MT-ND4 gene. The presence of this variant alone is insufficient to trigger disease symptoms, of which vision loss is the hallmark. Given the incomplete penetrance and inter-population variability in modifying factors, this study aimed to investigate two previously proposed genetic risk factors for LHON in the Polish population. Using quantitative PCR, we measured the mtDNA copy number in peripheral blood of affected and unaffected carriers of the m.11778G>A variant. In addition, we assessed the frequency of the PRICKLE3 c.157C>T variant in symptomatic, asymptomatic and control individuals using PCR-RFLP. Our results indicate that mtDNA copy number was not associated with LHON symptom manifestation in the Polish cohort under conditions tested, in contrast to previously reported associations in other populations. In addition, the PRICKLE3 c.157C>T variant was absent in our cohort, indicating that it is not a common modifier of LHON penetrance in the Polish population. These findings suggest that the incomplete penetrance of LHON in the Polish population may involve other modifying factors, such as yet unidentified nuclear DNA variants.

Journal of Applied Genetics
Poznan University of Medical Sciences (PL), Medical University of Warsaw (PL), Institute of Physiology and Pathology of Hearing (PL), Institute of Biochemistry and Biophysics, Polish Academy of Sciences (PL), Institute of Psychiatry and Neurology (PL), University of Warsaw (PL)
Uniwersytet Warszawski
Openalex Percentile: Top 11%
Mitochondrial Function and Pathology
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