Journal of Pediatric Neurology 2023; 21(05): 347-351
DOI: 10.1055/s-0043-1768473
Original Article

Microstructural Changes in Normal-Appearing White Matter in Pediatric X-Linked Adrenoleukodystrophy: A Diffusion Tensor Imaging Study

1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
2   Department of Radiology, Faculty of Medicine, Selcuk University, Konya, Türkiye
,
Jeffrey Rykken
1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
,
1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
,
Mark Folkertsma
1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
,
Yasemin Koksel
1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
,
Mehmet Gencturk
1   Division of Neuroradiology, Department of Radiology, University of Minnesota Medical Center, Minneapolis, Minnesota, United States
› Author Affiliations
Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors. H.C. was supported by a grant from The Scientific and Technical Research Council of Turkey (TUBITAK).

Abstract

Objective Diffusion tensor imaging (DTI) is an advanced magnetic resonance imaging (MRI) technique that provides quantitative diffusion data of water molecules in tissue. Usage of DTI in pediatric X-linked adrenoleukodystrophy (X-ALD) may adjunct earlier detection of brain involvement. This study aimed to analyze the normal-appearing white matter in pediatric patients with X-ALD using DTI.

Materials and Methods A retrospective database search was performed. Children with genetically diagnosed X-ALDs with a reportedly normal brain MRI between January 2010 and September 2021 were included. Fractional anisotropy (FA), radial diffusivity (RD), axial diffusivity (AD), and apparent diffusion coefficient were serially measured in 8 regions of the brain, and these measurements were compared with those in control patients.

Results The study group included 21 X-ALD patients and 11 control patients. The mean age was 6.64 ± 5.14 years. FA values of the genu and body of the corpus callosum were significantly lower in the ALD group than controls. There was also a statistically significant difference in FA, AD, and RD values of cerebellar white matter in the two groups.

Conclusion There is evidence of white matter damage on DTI in X-ALD patients with the normal-appearing white matter on conventional MRI sequences. The inclusion of DTI in routine MRI examinations of X-ALD patients may provide additional insight into possible white matter damage in that population.

Ethical Approval

For this article, no studies with human participants or animals were performed by any of the authors. All studies performed were in accordance with the ethical standards indicated in each case. The study was approved by the University of Minnesota Institutional Review Board.


Informed Consent

The requirement for a written informed consent was waived due to the retrospective nature of the study.




Publication History

Received: 16 February 2023

Accepted: 29 March 2023

Article published online:
22 May 2023

© 2023. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 
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