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@ARTICLE{ahin:282325,
author = {Şahin, Erdi and Samanci, Bedia and Yalçın Çakmaklı,
Gül and Lohmann, Ebba and Güven, Gamze and Gökalp, Ebru
Erzurumluoğlu and Gündüz, Ayşegül and Başak, Ayşe
Nazlı and Ertan, Sibel and Elibol, Bülent and Bilgiç,
Başar and Hanağası, Haşmet},
title = {{FBXO}7 {P}athogenic {V}ariants in {E}arly-{O}nset
{P}arkinsonism: {I}nsights from a {N}euroimaging
{P}erspective and {R}eview of the {L}iterature.},
journal = {Movement disorders clinical practice},
volume = {12},
number = {11},
issn = {2330-1619},
address = {New York, NY},
publisher = {Wiley},
reportid = {DZNE-2025-01286},
pages = {1972 - 1980},
year = {2025},
abstract = {Early onset parkinsonism caused by F-box only protein7
(FBXO7) pathogenic variants is a rare autosomal recessive
disorder presenting with a combination of parkinsonism,
pyramidal signs, dystonia, cognitive impairment, and
psychiatric features. Although previous case reports have
mentioned neuroimaging findings, there is no comprehensive
study characterizing the radiological spectrum of FBXO7
pathogenic variants.Ten patients from seven families
followed at two tertiary centers in Turkey were included.
The cohort included six males and four females with a mean
onset age of 21.1 years. Frontal atrophy was the most common
MRI finding, followed by global cortical and cerebellar
atrophy. One patient showed iron accumulation in the
pallidum, and two exhibited severe dopaminergic deficit on
DaTSCAN.Pathogenic variants in the FBXO7 gene have been
identified in diverse populations over the past 15 years,
contributing to a broader understanding of clinical and
radiological spectrum. Global cortical atrophy has emerged
as the most frequently reported neuroimaging finding in
FBXO7-related parkinsonism.FBXO7 pathogenic variants are
associated with heterogeneous neuroimaging findings. Frontal
and global cortical atrophy are common, while iron
deposition and DaTSCAN abnormalities offer additional
diagnostic clues. Larger, longitudinal studies are necessary
to establish specific imaging biomarkers for FBXO7-related
neurodegeneration.},
subtyp = {Review Article},
keywords = {Humans / Male / Female / Parkinsonian Disorders: genetics /
Parkinsonian Disorders: diagnostic imaging / Parkinsonian
Disorders: pathology / F-Box Proteins: genetics / Adult /
Neuroimaging / Young Adult / Magnetic Resonance Imaging /
Adolescent / Age of Onset / Atrophy / Brain: diagnostic
imaging / Brain: pathology / FBXO7 (Other) / early‐onset
(Other) / genetic (Other) / neuroimaging (Other) /
parkinsonism (Other) / FBXO7 protein, human (NLM Chemicals)
/ F-Box Proteins (NLM Chemicals)},
cin = {AG Gasser},
ddc = {610},
cid = {I:(DE-2719)1210000},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40740144},
pmc = {pmc:PMC12625086},
doi = {10.1002/mdc3.70269},
url = {https://pub.dzne.de/record/282325},
}