Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy
Abstract Objective Duchenne muscular dystrophy (DMD) is a neuromuscular disorder in which many patients also have neurobehavioral problems. Corticosteroids, the primary pharmacological treatment for DMD, have been shown to affect brain morphology in other conditions, but data in DMD are lacking. Thi...
| Main Authors: | , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
| Published: |
Wiley
2023-12-01
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| Series: | Annals of Clinical and Translational Neurology |
| Online Access: | https://doi.org/10.1002/acn3.51922 |
| _version_ | 1827583153908219904 |
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| author | Sam Geuens Jeroen Van Dessel Rosanne Govaarts Nadine A. Ikelaar Onno C. Meijer Hermien E. Kan Erik H. Niks Nathalie Goemans Jurgen Lemiere Nathalie Doorenweerd Liesbeth De Waele |
| author_facet | Sam Geuens Jeroen Van Dessel Rosanne Govaarts Nadine A. Ikelaar Onno C. Meijer Hermien E. Kan Erik H. Niks Nathalie Goemans Jurgen Lemiere Nathalie Doorenweerd Liesbeth De Waele |
| author_sort | Sam Geuens |
| collection | DOAJ |
| description | Abstract Objective Duchenne muscular dystrophy (DMD) is a neuromuscular disorder in which many patients also have neurobehavioral problems. Corticosteroids, the primary pharmacological treatment for DMD, have been shown to affect brain morphology in other conditions, but data in DMD are lacking. This study aimed to investigate the impact of two corticosteroid regimens on brain volumetrics in DMD using magnetic resonance imaging (MRI). Methods In a cross‐sectional, two‐center study, T1‐weighted MRI scans were obtained from three age‐matched groups (9–18 years): DMD patients treated daily with deflazacort (DMDd, n = 20, scan site: Leuven), DMD patients treated intermittently with prednisone (DMDi, n = 20, scan site: Leiden), and healthy controls (n = 40, both scan sites). FSL was used to perform voxel‐based morphometry analyses and to calculate intracranial, total brain, gray matter, white matter, and cerebrospinal fluid volumes. A MANCOVA was employed to compare global volumetrics between groups, with site as covariate. Results Both patient groups displayed regional differences in gray matter volumes compared to the control group. The DMDd group showed a wider extent of brain regions affected and a greater difference overall. This was substantiated by the global volume quantification: the DMDd group, but not the DMDi group, showed significant differences in gray matter, white matter, and cerebrospinal fluid volumes compared to the control group, after correction for intracranial volume. Interpretation Volumetric differences in the brain are considered part of the DMD phenotype. This study suggests an additional impact of corticosteroid treatment showing a contrast between pronounced alterations seen in patients receiving daily corticosteroid treatment and more subtle differences in those treated intermittently. |
| first_indexed | 2024-03-08T23:01:44Z |
| format | Article |
| id | doaj.art-add3ba9314e94a10b739bcd5d963f0ee |
| institution | Directory Open Access Journal |
| issn | 2328-9503 |
| language | English |
| last_indexed | 2024-03-08T23:01:44Z |
| publishDate | 2023-12-01 |
| publisher | Wiley |
| record_format | Article |
| series | Annals of Clinical and Translational Neurology |
| spelling | doaj.art-add3ba9314e94a10b739bcd5d963f0ee2023-12-15T16:39:24ZengWileyAnnals of Clinical and Translational Neurology2328-95032023-12-0110122324233310.1002/acn3.51922Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophySam Geuens0Jeroen Van Dessel1Rosanne Govaarts2Nadine A. Ikelaar3Onno C. Meijer4Hermien E. Kan5Erik H. Niks6Nathalie Goemans7Jurgen Lemiere8Nathalie Doorenweerd9Liesbeth De Waele10Child Neurology University Hospitals Leuven Leuven BelgiumDepartment of Neurosciences, Center for Developmental Psychiatry UPC‐KU Leuven Leuven BelgiumC.J. Gorter MRI Center, Radiology Leiden University Medical Center Leiden NetherlandsDuchenne Center Netherlands Leiden NetherlandsDepartment of Medicine Leiden University Medical Center Leiden NetherlandsC.J. Gorter MRI Center, Radiology Leiden University Medical Center Leiden NetherlandsDuchenne Center Netherlands Leiden NetherlandsChild Neurology University Hospitals Leuven Leuven BelgiumPediatric Hemato‐Oncology University Hospitals Leuven Leuven BelgiumC.J. Gorter MRI Center, Radiology Leiden University Medical Center Leiden NetherlandsChild Neurology University Hospitals Leuven Leuven BelgiumAbstract Objective Duchenne muscular dystrophy (DMD) is a neuromuscular disorder in which many patients also have neurobehavioral problems. Corticosteroids, the primary pharmacological treatment for DMD, have been shown to affect brain morphology in other conditions, but data in DMD are lacking. This study aimed to investigate the impact of two corticosteroid regimens on brain volumetrics in DMD using magnetic resonance imaging (MRI). Methods In a cross‐sectional, two‐center study, T1‐weighted MRI scans were obtained from three age‐matched groups (9–18 years): DMD patients treated daily with deflazacort (DMDd, n = 20, scan site: Leuven), DMD patients treated intermittently with prednisone (DMDi, n = 20, scan site: Leiden), and healthy controls (n = 40, both scan sites). FSL was used to perform voxel‐based morphometry analyses and to calculate intracranial, total brain, gray matter, white matter, and cerebrospinal fluid volumes. A MANCOVA was employed to compare global volumetrics between groups, with site as covariate. Results Both patient groups displayed regional differences in gray matter volumes compared to the control group. The DMDd group showed a wider extent of brain regions affected and a greater difference overall. This was substantiated by the global volume quantification: the DMDd group, but not the DMDi group, showed significant differences in gray matter, white matter, and cerebrospinal fluid volumes compared to the control group, after correction for intracranial volume. Interpretation Volumetric differences in the brain are considered part of the DMD phenotype. This study suggests an additional impact of corticosteroid treatment showing a contrast between pronounced alterations seen in patients receiving daily corticosteroid treatment and more subtle differences in those treated intermittently.https://doi.org/10.1002/acn3.51922 |
| spellingShingle | Sam Geuens Jeroen Van Dessel Rosanne Govaarts Nadine A. Ikelaar Onno C. Meijer Hermien E. Kan Erik H. Niks Nathalie Goemans Jurgen Lemiere Nathalie Doorenweerd Liesbeth De Waele Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy Annals of Clinical and Translational Neurology |
| title | Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy |
| title_full | Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy |
| title_fullStr | Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy |
| title_full_unstemmed | Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy |
| title_short | Comparison of two corticosteroid regimens on brain volumetrics in patients with Duchenne muscular dystrophy |
| title_sort | comparison of two corticosteroid regimens on brain volumetrics in patients with duchenne muscular dystrophy |
| url | https://doi.org/10.1002/acn3.51922 |
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