Evaluation of Mitochondrial Complex 1 Density with [18F]BCPP-EF in a Murine Model and Individuals with Friedreich Ataxia Article Swipe
Laigao Chen
,
Gaia Rizzo
,
Christine Bulawa
,
Koene R. A. Van Dijk
,
Erica C. Henning
,
Alain Martelli
,
Jeffrey Palmer
,
Avery McIntosh
,
Marko J. Pregel
,
Pengling Sun
,
Emmanuel Adewunmi
,
M C Aldridge
,
Jackson Chan
,
Roger N. Gunn
,
Mickaël Huiban
,
Allan Listanco
,
Peter T. Loudon
,
Sara Moz
,
Jan Passchier
,
L R Sauvage
,
R. J. Stewart
,
Lisa Wells
,
Eugenii A. Rabiner
,
Lawrence Charnas
,
Richard Festenstein
·
YOU?
·
· 2025
· Open Access
·
· DOI: https://doi.org/10.2967/jnumed.124.268698
YOU?
·
· 2025
· Open Access
·
· DOI: https://doi.org/10.2967/jnumed.124.268698
Friedreich ataxia is caused by mutations in the frataxin gene, leading to neurodegeneration and premature death from cardiac dysfunction. Loss of frataxin impacts mitochondrial complex 1 (MC1) activity, suggesting MC1 may be a potential biomarker of frataxin levels and function. Biomarkers evaluated by noninvasive techniques are needed to monitor disease progression and treatment effects in people with Friedreich ataxia. Methods: PET with [18F]BCPP-EF, a ligand with high binding specificity for MC1, was used to measure cardiac and brain MC1 density in a mouse model of Friedreich ataxia and in healthy volunteers and participants with Friedreich ataxia. Results: An imaging protocol was developed in humans that included a 70-min brain scan immediately after administration of [18F]BCPP-EF followed by a 60-min cardiac scan 255 min after [18F]BCPP-EF administration. Cardiac [18F]BCPP-EF binding in participants with Friedreich ataxia was lower than that in healthy volunteers and in a mouse model of Friedreich ataxia versus wild-type mice (∼50% reduction in both). In the brain, no statistically significant difference in the [18F]BCPP-EF binding was detected between participants with Friedreich ataxia and healthy volunteers. Correlation analyses showed that blood frataxin and cardiac [18F]BCPP-EF levels decreased with increasing guanine-adenine-adenine expansion size (R = -0.82 and -0.78, respectively; both P < 0.05) but not in the precentral gyrus (R = 0.63; P < 0.05). Conclusion: MC1 density as measured using [18F]BCPP-EF-based PET may be a viable biomarker of mitochondrial deficit and frataxin levels in people with Friedreich ataxia.
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Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.2967/jnumed.124.268698
- https://jnm.snmjournals.org/content/jnumed/early/2025/07/31/jnumed.124.268698.full.pdf
- OA Status
- hybrid
- References
- 28
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4412806252
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
-
https://openalex.org/W4412806252Canonical identifier for this work in OpenAlex
- DOI
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https://doi.org/10.2967/jnumed.124.268698Digital Object Identifier
- Title
-
Evaluation of Mitochondrial Complex 1 Density with [18F]BCPP-EF in a Murine Model and Individuals with Friedreich AtaxiaWork title
- Type
-
articleOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2025Year of publication
- Publication date
-
2025-07-31Full publication date if available
- Authors
-
Laigao Chen, Gaia Rizzo, Christine Bulawa, Koene R. A. Van Dijk, Erica C. Henning, Alain Martelli, Jeffrey Palmer, Avery McIntosh, Marko J. Pregel, Pengling Sun, Emmanuel Adewunmi, M C Aldridge, Jackson Chan, Roger N. Gunn, Mickaël Huiban, Allan Listanco, Peter T. Loudon, Sara Moz, Jan Passchier, L R Sauvage, R. J. Stewart, Lisa Wells, Eugenii A. Rabiner, Lawrence Charnas, Richard FestensteinList of authors in order
- Landing page
-
https://doi.org/10.2967/jnumed.124.268698Publisher landing page
- PDF URL
-
https://jnm.snmjournals.org/content/jnumed/early/2025/07/31/jnumed.124.268698.full.pdfDirect link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
hybridOpen access status per OpenAlex
- OA URL
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https://jnm.snmjournals.org/content/jnumed/early/2025/07/31/jnumed.124.268698.full.pdfDirect OA link when available
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Ataxia, Endocrinology, Chemistry, Internal medicine, Neuroscience, Psychology, MedicineTop concepts (fields/topics) attached by OpenAlex
- Cited by
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0Total citation count in OpenAlex
- References (count)
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28Number of works referenced by this work
- Related works (count)
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10Other works algorithmically related by OpenAlex
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