FMRP regulates MFF translation to locally direct mitochondrial fission in neurons

Fragile X messenger ribonucleoprotein (FMRP) is a critical regulator of translation, whose dysfunction causes fragile X syndrome. FMRP dysfunction disrupts mitochondrial health in neurons, but it is unclear how FMRP supports mitochondrial homoeostasis. Here we demonstrate that FMRP granules are recruited to the mitochondrial midzone, where they mark mitochondrial fission sites in axons and dendrites. Endolysosomal vesicles contribute to FMRP granule positioning around mitochondria and facilitate FMRP-associated fission via Rab7 GTP hydrolysis. Cryo-electron tomography and real-time translation imaging reveal that mitochondria-associated FMRP granules are ribosome-rich structures that serve as sites of local protein synthesis. Specifically, FMRP promotes local translation of mitochondrial fission factor (MFF), selectively enabling replicative fission at the mitochondrial midzone. Disrupting FMRP function dysregulates mitochondria-associated MFF translation and perturbs fission dynamics, resulting in increased peripheral fission and an irregular distribution of mitochondrial nucleoids. Thus, FMRP regulates local translation of MFF in neurons, enabling precise control of mitochondrial fission.

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Publication Details

Journal
Nature Cell Biology
Published
2024-11-15
DOI
https://doi.org/10.1038/s41556-024-01544-2
Citations
33
Primary Topic
Genetics and Neurodevelopmental Disorders
Type
article
Field-Weighted Citation Impact
18.16

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article

FMRP regulates MFF translation to locally direct mitochondrial fission in neurons

Siewert Hugelier, Yi‐Wei Chang, Thomas A Jongens, Charles Bond et al.
33 citations
Nature Cell Biology
Genetics and Neurodevelopmental Disorders
18.16
article

FMRP regulates MFF translation to locally direct mitochondrial fission in neurons

Siewert Hugelier, Yi‐Wei Chang, Thomas A Jongens, Charles Bond, Adam R. Fenton, Ruchao Peng, Melike Lakadamyali, Erika L.F. Holzbaur
article en
33 citations

Abstract

Fragile X messenger ribonucleoprotein (FMRP) is a critical regulator of translation, whose dysfunction causes fragile X syndrome. FMRP dysfunction disrupts mitochondrial health in neurons, but it is unclear how FMRP supports mitochondrial homoeostasis. Here we demonstrate that FMRP granules are recruited to the mitochondrial midzone, where they mark mitochondrial fission sites in axons and dendrites. Endolysosomal vesicles contribute to FMRP granule positioning around mitochondria and facilitate FMRP-associated fission via Rab7 GTP hydrolysis. Cryo-electron tomography and real-time translation imaging reveal that mitochondria-associated FMRP granules are ribosome-rich structures that serve as sites of local protein synthesis. Specifically, FMRP promotes local translation of mitochondrial fission factor (MFF), selectively enabling replicative fission at the mitochondrial midzone. Disrupting FMRP function dysregulates mitochondria-associated MFF translation and perturbs fission dynamics, resulting in increased peripheral fission and an irregular distribution of mitochondrial nucleoids. Thus, FMRP regulates local translation of MFF in neurons, enabling precise control of mitochondrial fission.

Nature Cell BiologyVol. 26(12)
University of Pennsylvania (US)
David and Lucile Packard Foundation, National Institutes of Health
Zero hunger
Openalex Percentile: Top 0%
Genetics and Neurodevelopmental Disorders
18.16
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FMRP regulates MFF translation to locally direct mitochondrial fission in neurons — Siewert Hugelier, Yi‐Wei Chang, et al. · Nature Cell Biology (2024) | TGRS Research Map | TGRS