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@ARTICLE{Sardhara:1052293,
author = {Sardhara, Trushal and Dadsena, Ravi and Aydin, Roland C.
and Hilgers, Ralf-Dieter and Horn, Leon and Schulz, Jörg B.
and Reetz, Kathrin and Romanzetti, Sandro and Dogan, Imis
and Lischewski, Stella A. and Konrad, Kerstin and Pishnamaz,
Miguel and Praster, Maximillian and Clavel, Thomas and
Jankowski, Vera and Jankowski, Joachim and Pabst, Oliver and
Marx-Schütt, Katharina and Marx, Nikolaus and Möllmann,
Julia and Jacobsen, Malte and Dukart, Juergen and Eickhoff,
Simon},
title = {{D}eep learning-based 3{D} reconstruction of dentate nuclei
in {F}riedreich’s ataxia from {T}2*weighted {MR} images},
journal = {Machine learning with applications},
volume = {22},
issn = {2666-8270},
address = {Amsterdam},
publisher = {Elsevier},
reportid = {FZJ-2026-00910},
pages = {100790 -},
year = {2025},
abstract = {Dentate nucleus (DN) degeneration is a key
neuropathological feature in Friedreich’s ataxia (FRDA),
and its accurate quantification is critical for
understanding disease progression. However, its
visualization and volumetry require iron-sensitive imaging
techniques and time-consuming segmentation procedures,
posing challenges for conventional ML approaches due to
small datasets typical of rare diseases. We present a
transfer learning–based machine learning pipeline for
automated DN segmentation that directly uses standard
T2*-weighted Magnetic Resonance Imaging (MRI), which
highlights the DN without additional processing, and is
designed to perform robustly with limited annotated data.
Using 38 manually labeled subjects (18 FRDA, 20 controls),
the model was validated via five-fold cross-validation and
an independent hold-out test set, achieving Dice scores of
0.81–0.87 and outperforming classical atlas-based methods.
Pretraining improved performance by $∼10\%$ in patients
and $>5\%$ in controls. Applied to 181 longitudinal scans
from 33 FRDA patients and 33 controls, the model revealed
significantly reduced DN volumes in FRDA, with reductions
correlating with disease duration and clinical severity over
time. Our approach provides a scalable and reproducible
segmentation framework, requiring minimal annotated data and
no preprocessing, while demonstrating robust performance
across cross-validation and independent testing.
Additionally, it enables the first longitudinal volumetric
analysis of DN in FRDA using standard T2*-weighted MRI,
demonstrating its practical utility for monitoring
neurodegenerative changes. Overall, this work illustrates
how transfer learning can overcome data scarcity in rare
diseases and provides a robust methodology for automated MRI
segmentation in both research and clinical applications.},
cin = {INM-7},
ddc = {004},
cid = {I:(DE-Juel1)INM-7-20090406},
pnm = {5251 - Multilevel Brain Organization and Variability
(POF4-525)},
pid = {G:(DE-HGF)POF4-5251},
typ = {PUB:(DE-HGF)16},
doi = {10.1016/j.mlwa.2025.100790},
url = {https://juser.fz-juelich.de/record/1052293},
}