EMBO/SourceData huggingface dataset:
Figure legend: "(C) Chop and Chac1 transcripts are upregulated in E14 Elp3cKO compared to WT cochleae (n=8/6 for WT/KO; unpaired t-test two-tailed; p=0.0001/0.0015/0.0010; t=9.535/4.090/4.307; DF=12/12/12 for Elp3, Chop and Chac1 respectively; mean±SEM presented) indicating that the pro-apoptotic arm of the unfolded protein response (UPR) is activated in Elp3-deficient SGNs."
NCBI gene IDs:
"Chac1: 69065
Chop: 13198
Elp3: 74195"
Figure legend: "(D-E) Proteostat staining of whole-mounted P0 organs of Corti reveal the presence of aggregates in HCs upon loss of Elp3. Orthogonal slices (Orthog) reveal aggregated proteins localize at the apical surface of Elp3cKO HCs. Scale bars =5µm. #IHC; *OHC1; **OHC2; ***OHC3."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(F) Protein extracts from P1 cochleae show increased levels of polyubiquitinated conjugates upon loss of Elp3, confirming proteostasis disruption."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(G) The levels of Chop and Chac1 transcripts are unaffected by Elp3 depletion in P0 organs of Corti (n=3 animals per genotype; unpaired t-test two-tailed; p=0.0003/0.4296/0.056; t=11.62/0.8778/2.662; DF=4/4/4 for Elp3, Chop and Chac1 respectively; mean±SEM presented) suggesting that proteostasis disruption in Elp3cKO HCs is not sufficient to induce UPR-mediated apoptosis."
NCBI gene IDs:
"Chac1: 69065
Chop: 13198
Elp3: 74195"
Figure legend: "(A) Scanning electron micrographs of cochlear tissue from P19 animals indicate no HC loss in the absence of Elp3 but illustrate some defects of polarity, as misaligned HCs and dysmorphic stereociliary bundles are visible (red arrows and white arrowheads, respectively). Scale bars=5µ"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(B) Surface view of P1 actin-stained organ of Corti confirms defective establishment of polarity in Elp3cKO as numerous stereociliary bundles are misaligned along the medio-lateral axis (M-L) or misshaped. Scale bars=5µm. Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(C) Elp3cKOs exhibit a broader distribution of hair cell orientation. The angles of deviation between the stereociliary bundle and the medio-lateral axis are plotted in rose diagrams, for each OHC row. Whereas the majority of WT bundles are pointing to the most lateral region of the tissue (within a range of 10°), many Elp3cKO bundles of the two last rows of OHCs present increased angles of deviation (WT/Elp3cKO OHC1: n=99/99, p>0.1, DF=99; OHC2: n=99/100, p&lt;0.001, DF=99; OHC3: n=99/98, p&lt;0.001, DF=98; pooled data from 6 animals per genotype; Watson U2 test). Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "Elp3cKOs HCs harbour a flatter stereociliary bundle that is centrally shifted. (D) Scanning electron micrographs of HCs from P1 WT and Elp3cKO cochleae (scale bar=2µm) illustrating the misshaped stereociliary bundles and the shorter kinocilium (false coloured in pink) of Elp3cKO HCs. Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "Elp3cKOs HCs harbour a flatter stereociliary bundle that is centrally shifted. (E) Graph presents the mean angle of HC bundle (n=6 animals; one-way ANOVA, Tukey's multiple comparisons test; ***p&lt;0.001, F=13.69, DF=7; mean±SD)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "Elp3cKOs HCs harbour a flatter stereociliary bundle that is centrally shifted. (F) Elp3cKO HCs possess a significantly larger bare zone (WT/KO IHC: n= 23/24 cells; OHC1: n=27/27; OHC2: n=27/29; OHC3: n=30/29; cumulative data collected from 3 animals per genotype; One-way ANOVA, Tukey's multiple comparisons test; ***p&lt;0.001, *p&lt;0.05 F=16.72, DF=7; mean±SD)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "Elp3cKOs HCs harbour a flatter stereociliary bundle that is centrally shifted. (G) Elp3cKO HCs possess a significantly smaller medial zone (WT/KO IHC: n= 23/24 cells; OHC1: n=27/27; OHC2: n=27/29; OHC3: n=27/29; cumulative data collected from 3 animals per genotype; One-way ANOVA, Tukey's multiple comparisons test; ***p&lt;0.001, *p&lt;0.05, F=63.3, DF=7; mean±SD)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(H) Kinocilia are shorter in Elp3cKO HCs (WT/KO IHC: n=15/15; OHC1: n=30/32; OHC3: n=31/30; OHC3: n=27/30; data collected from 3 animals per genotype; One-way ANOVA, Tukey's multiple comparisons test, ***p&lt;0.001, **p&lt;0.01F=19.6, DF=7; mean±SD)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(I) High magnification of representative HCs from P1 WT and Elp3cKO cochlea stained for F-actin and gamma-tubulin (white). The basal body of kinocilia is shifted centrally at the apical surface of Elp3cKO HCs. Scale bars=2µm. Polar plotting of kinocilium basal body position for each row of HCs of WT and Elp3cKO (WT/KO IHC: n= 42/32; OHC1: n= 47/48; OHC2: n= 47/45; OHC3: n= 44/44; cumulative data collected from 3 animals per genotype). Cellular orientations were tested using Watson U2 test (p&lt;0.001 between WT/Elp3cKO, for all HC rows) and the mean vector lengths, determined from the cell centre to the basal body, were compared using t-tests (p&lt;0.001 between WT/Elp3cKO, for all HC rows)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "The localization of apical polarity proteins is affected by Elp3 loss. Surface views of mid-basal turn of P1 cochleae from WT or Elp3cKO mice stained with LGN (J) and F-actin. The enrichment of these apical polarity regulators at the lateral edges of HCs is disrupted in Elp3cKO HCs. Scale bars=5µm (upper panels) and 2µm (lower panels). Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "The localization of apical polarity proteins is affected by Elp3 loss. Surface views of mid-basal turn of P1 cochleae from WT or Elp3cKO mice stained with Gαi3 (K) and F-actin. The enrichment of these apical polarity regulators at the lateral edges of HCs is disrupted in Elp3cKO HCs. Scale bars=5µm (upper panels) and 2µm (lower panels). Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "The localization of apical polarity proteins is affected by Elp3 loss. Surface views of mid-basal turn of P1 cochleae from WT or Elp3cKO mice stained with aPKC (L) and F-actin. The enrichment of these apical polarity regulators at the medial (L) edges of HCs is disrupted in Elp3cKO HCs. Scale bars=5µm (upper panels) and 2µm (lower panels). Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(A-B) Polarized enrichment of LGN at the cortices of dividing HEK293T cells is disrupted upon Elp3 or Alkbh8 knockdown. (A) HEK293T cells transfected with siRNAs for Elp3, Alkbh8 or control and immunolabelled with LGN, acetylated alpha-tubulin and DAPI. Scale bar=5µm. (B) The mean fluorescence intensity profile of LGN across the radii perpendicular to division in HEK293T cells was quantified (siRNA Control/Elp3/Alkbh8: n=27/30/27; pooled from 3 independent experiments; mean±SEM)."
NCBI gene IDs:
"Alkbh8: 91801
Elp3: 55140"
Figure legend: "(C-D) The length of the primary cilium is reduced in non-dividing HEK293T cells upon Elp3 or Albh8 knockdown. (C) Control, Elp3 or Alkbh8 siRNA-treated HEK293T cells were immunolabelled for basal body marker ALMS1, axoneme marker Arl13b and DAPI. Scale bar=5µm. (D) Primary cilium length of non-dividing HEK293T was measured and pooled data were plotted (siRNA Control/Elp3/Alkbh8: n=94/81/90; data collected from 3 independent experiments; One-way ANOVA, Tukey's multiple comparisons test, ***p&lt;0.001, F=21.46, DF=4; minimum, first quartile, median, third quartile and maximum presented)."
NCBI gene IDs:
"Albh8: 91801
Alkbh8: 91801
Elp3: 55140"
Figure legend: "(E-F) Elp3 knockdown increases the amount of polyubiquitinated conjugates in HEK293T cells (n=3; One-way ANOVA, Tukey's multiple comparisons test, **p&lt;0.01, *p&lt;0.05, F=16.51, DF=2; mean±SD)."
NCBI gene IDs:
"Elp3: 55140"
Figure legend: "(G) Increased formation of aggresome-like structures in Elp3- or Alkbh8-depleted HEK293T. Aggresomes (red); DAPI (blue). Enlarged images depict examples of non-dividing (left) and dividing (right) cells. Scale bar=10µm."
NCBI gene IDs:
"Alkbh8: 91801
Elp3: 55140"
Figure legend: "LGN and Gαi3 protein levels are not affected by Elp3 loss. (A) Western blots with total lysates from control and Elp3 siRNA-treated HEK293T cells and corresponding quantifications of normalized LGN, Gαi3 and Elp3 levels (n=6 from 2 independent cultures; Unpaired two-tailed t-test, LGN: p=0.749, t=0.328, DF=10, Gαi3: p=0.275, t=1.155, DF=10, Elp3: ***p=0.0003, t=5.438, DF=10, mean±SEM)."
NCBI gene IDs:
"Elp3: 55140"
Figure legend: "LGN and Gαi3 protein levels are not affected by Elp3 loss. (B) Western blots on P0 WT and Elp3cKO cochlear extracts and corresponding quantifications of normalized LGN and Gαi3 levels (n=3, each lysate obtained from cochleae pooled from 2 animals; Unpaired two-tailed t-test, LGN: p=0.586, t=0.591, DF=4, Gαi3: p=0.963, t=0.0491, DF=4, mean±SEM)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(C) Mislocalized LGN and Gαi3 are not found in protein aggregates formed upon Elp3 depletion. HEK293T transfected with Elp3 siRNA were co-stained for aggresomes, Dapi and LGN or Gαi3. Scale bar=5µm."
NCBI gene IDs:
"Elp3: 55140"
Figure legend: "Microtubular transport speed is reduced by the presence of aggresome-like structures in HEK293T cells and is improved upon 4-PBA treatment. The mean velocity of lysosomes was measured on time-lapse recordings of HEK293T cells labelled with LysoTracker®. (E): siCtl/+4PBA n=622/677; siElp3/+4PBA n=674/577; siAlkbh8/+4PBA n=460/484, pooled from 4 experiments, ***p&lt;0.001. All data analysed with Kruskal-Wallis ANOVA, Dunn's multiple comparisons test. Box/whisker plots depict minimum, first quartile, median, third quartile, and maximum."
NCBI gene IDs:
"Alkbh8: 91801
Elp3: 55140"
Figure legend: "Microtubular transport speed is reduced by the presence of aggresome-like structures in cultured HCs, and is improved upon 4-PBA treatment. The mean velocity of lysosomes was measured on time-lapse recordings of HCs, at the level of their apical surface labelled with LysoTracker®. (G): WT/WT+4PBA/Elp3cKO/Elp3cKO+4PBA n=732/763/730/738, pooled from 4 OCs per condition, ***p&lt;0.001, **p&lt;0.01. All data analysed with Kruskal-Wallis ANOVA, Dunn's multiple comparisons test. Box/whisker plots depict minimum, first quartile, median, third quartile, and maximum."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "In vivo administration of 4-PBA improves LGN localization and kinocilium position in Elp3cKO cochlear HCs. Pregnant mice were treated daily with saline or 4-PBA from E13.5 and the cochleae of WT and Elp3cKO embryos were analysed by immunostainings at E17.5. (D) The arc of lateral LGN enrichment remains in many Elp3cKO HCs after 4PBA administration. Scale bars=5µm; magnified views=2µm. Data information: #IHC; *OHC1; **OHC2; ***OHC3"
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "In vivo administration of 4-PBA improves LGN localization and kinocilium position in Elp3cKO cochlear HCs. Pregnant mice were treated daily with saline or 4-PBA from E13.5 and the cochleae of WT and Elp3cKO embryos were analysed by immunostainings at E17.5. (E) Acetylated alpha-tubulin immunostaining showing kinocilium position in WT or Elp3cKO OCs in the presence or absence of 4-PBA (yellow arrowheads = base of kinocilium; scale bar=2µm)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "In vivo administration of 4-PBA improves LGN localization and kinocilium position in Elp3cKO cochlear HCs. Pregnant mice were treated daily with saline or 4-PBA from E13.5 and the cochleae of WT and Elp3cKO embryos were analysed by immunostainings at E17.5. (F) Kinocilium position plotted as a fraction of HC diameter for each HC row (WT/WT+4PBA/Elp3cKO/Elp3cKO+4PBA IHC: n=35/31/35/36, p=0.6812, F=4.510, DF=3; OHC1: n=36/32/37/40, ***p&lt;0.001, **p&lt;0.01, F=27.60, DF=3; OHC2: n=35/33/38/42, ***p&lt;0.001, F=76.62, DF=3; OHC3: n=36/32/38/39, ***p&lt;0.001, *p&lt;0.05, F=27.86, DF=3; pooled from 3 animals per condition; mean±SD; One-way ANOVA with Tukey's multiple comparisons test)."
NCBI gene IDs:
"Elp3: 74195"
Figure legend: "(A) Representative images (and details from boxed regions) of WT MEF  cells transfected with   YFPParkin (green) and treated with FCCP (10 μM) to induce mitophagy. Cells were immunostained with anti-Tom20 (magenta) to reveal the mitochondrial network. Each row represents one stage in the process of  mitophagy that has been used to score the progression of mitophagy at different  time-points for the different conditions tested (the specific time-point of each example is indicated in the  image). From top to bottom: i) \"Diffuse Parkin\" defines cells in where  Parkin expression is homogenously distributed throughout the cytoplasm (quantified in B). ii) Soon after  mitochondrial  damage  Parkin appears enriched in small \"puncta\" throughout the cytoplasm localizing within isolated mitochondria (quantified in C). iii) Later in the process these puncta structures aggregate in bigger structures accumulating in perinuclear  regions (quantified in D). iv) The process of Parkin translocation eventually affects all the mitochondrial population and usually collapses in few large structures possibly forming part of big autolysosomes in the perinuclear region (quantified in D). Scoring was performed from 3 independent experiments (n=3; one way ANOVA with Dunnett post test). Error bars represent s.e.m. Significance: *p&lt;0.05 and **p&lt;0.01"
NCBI gene IDs:
"YFP: 
Parkin: 5071"
Figure legend: "(F) Representative images (and details from boxed regions) of WT MEFs cells transfected with GFPPINK1 (green) and treated with FCCP (10 μM) to induce  mitophagy. Cells were immunostained  with anti-Tom20 (magenta) to reveal the mitochondrial  network. Each row represents one of the three categories of  GFP signal used to describe  PINK1 translocation to  mitochondria after mitochondrial damage (cytoplasmic, intermediate and mitochondrial). The scoring of these categories was used to describe PINK1 translocation to mitochondria in WT and  MiroDKO cells at different time-points shortly after mitochondrial insult (G). Data collected from 3 independent experiments (n=3). Error bars represent s.e.m. Significance: *p&lt;0.05 and **p&lt;0.01 "
NCBI gene IDs:
"GFP: 
PINK1: 65018
Miro: 214952///59040"
Figure legend: "B) Representative  images showing full length control and selected Miro1 lysine mutants (Miro15R, Miro1allR and Miro1R572K) expressing in MiroDKO MEF  cells. All constructs (myc-tag, green) localise to mitochondria (Tom20, red)."
NCBI gene IDs:
"Miro1: 55288
Miro: 214952///59040"
Figure legend: "C) Immunoblot showing efficient expression of  Miro1 lysine mutants in   FlagParkin expressing SH-SY5Y cells."
NCBI gene IDs:
"Miro1: 55288"
Figure legend: "D) Ubiquitination assay showing that Miro1 lysine mutants have reduced ubiquitination upon  FCCP treatment (1h, 10 μM) in FlagParkin overexpressing SH-SY5Y. The effect is quantified in (E) (n=4 independent experiments for Miro1WT and Miro15R and n=3 for  Miro1allR and Miro1R572K, ANOVA with Sidak's post hoc test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001."
NCBI gene IDs:
"Flag: 
Parkin: 5071
Miro1: 55288"
Figure legend: "F) Representative western blot showing a degradation assay in FlagParkin overexpressing SH-SY5Y  cells transfected with  Miro1WT, Miro15R or Miro1allR constructs and treated with FCCP  (10 μM) for 3 or 6 hours. Quantification of Miro1 levels (G) the matrix protein PDH E1α in (H) and  Parkin levels (I) (n=4 independent experiments; ANOVA with Sidak's post hoc test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001. "
NCBI gene IDs:
"Flag: 
Parkin: 5071
Miro1: 55288"
Figure legend: "(A) Representative images at 3 hours of FCCP treatment (10 μM) of YFPParkin  (green) expressing  MiroDKO cells co-transfected with the specified myc-tagged versions of Miro1 (cyan). Tom20 (red) was used to reveal the mitochondrial network. A detail of YFPParkin translocation onto the mitochondrial  network is also shown for each condition."
NCBI gene IDs:
"myc: 
YFP: 
Parkin: 5071
Miro: 214952///59040"
Figure legend: "(B and C) Quantification of the  fraction of cells showing advanced stages of the mitophagic process  (cells showing mitochondrial aggregation due to advanced  Parkin translocation or the complete translocation of  Parkin onto all the mitochondrial  network) at 1 hour (B) or 3 hours (C) after  FCCP treatment (data collected from at least 3 independent experiments: WT n=7,  MiroDKO n=8, +mycMiro1WT=7, mycMiro15R=3, mycMiro1allR=3; mycMiro2=3; one way ANOVA with Dunnett post test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001"
NCBI gene IDs:
"myc: 
Miro1: 55288
Miro2: 214952
Miro: 214952///59040"
Figure legend: "D) Western blots and quantification of  Miro1 degradation in MiroDKO cells co-expressing YFPParkin and the indicated  myc-tagged Miro1 constructs and treated with  FCCP (10 μM) for the indicated times  (n=3 different experiments; 2-way-ANOVA with Dunnett post test; comparisons between Miro1WT and Miro1allR are represented with yellow asteriscs, comparisons between Miro1WT and Miro15R are represented in green and comparisons between Miro15R and Miro1allR are represented in black)."
NCBI gene IDs:
"myc: 
YFP: 
Parkin: 5071
Miro1: 55288
Miro: 214952///59040"
Figure legend: "E). Representative  images at 24 hours of FCCP treatment (10 μM) of YFPParkin (green) expressing MiroDKO cells co-transfected with the specified myc-tagged versions of Miro1 (cyan). Tom20 (red) was used to reveal the  mitochondrial  network. Expression of both ubiquitin mutants (Miro15R and Miro1allR - arrowheads) protects from the mitophagic clearance of  mitochondria  (arrows) after damage. (F) Quantification of the fraction  of cells showing the complete loss of  Tom20 signal of mitochondria  after 24 hours of FCCP  treatment (data collected from at least 3 independent experiments: MiroDKO n=8, mycMiro1WT=7,   mycMiro15R=3, mycMiro1allR=3; one way ANOVA with Dunnett post test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001 "
NCBI gene IDs:
"myc: 
YFP: 
Parkin: 5071
Miro1: 55288
Miro: 214952///59040"
Figure legend: "(A) Representative  confocal images of the soma of WT and Miro1KO cortical neurons expressing YFPParkin and MtDsRed  after valinomycin treatment at the indicated  time points (scale bars = 5 µm)."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(B) Airyscan confocal images  of Parkin recruitment after 5 hours of valinomycin treatment in WT and Miro1KO neuronal  processes (scale bars = 2.5 µm)."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(C)  Fluorescent linescans of YFPParkin and MtDsRed signal in WT and Miro1KO neurons after 5 hours of valinomycin treatment (lines shown in A)."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(D) Quantification of Parkin recruitment to mitochondria (YFPParkin signal overlapping MtDsRed  signal - intensity-adjusted and normalised to t=0) in WT and Miro1KO neurons following valinomycin treatment (n=15 cells all conditions per genotype over 3 neuronal preparations; two-way ANOVA). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001"
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(E) Quantification of mitochondrial   occupancy (area of MtDsRed signal in soma / entire area of soma) in the somas of WT and Miro1KO neurons following  valinomycin treatment (n=15 cells all conditions per genotype over 3 neuronal preparations; two-way ANOVA)."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(F) Quantification of mitochondrial   clearance in  somas of WT and Miro1KO neurons between 2 and 5 hours of valinomycin treatment (n=15 cells all conditions per genotype over 3  neuronal preparations; Unpaired t-test)."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(G) Representative confocal images of WT,  Miro1KO and PINK1KO cortical neurons immunostained  with MAP2  (cyan) and s65-phospho-ubiquitin (green) after valinomycin treatment (Scale bars = 20µm). (H) Quantification of s65-phospho-ubiquitin signal  intensity within  MAP2 signal (normalised to MAP2 area and t=0) in Miro1WT, Miro1KO and PINK1KO neurons following valinomycin treatment n= 3, 4 and 3  embryos from WT, Miro1KO and PINK1KO embryos, respectively (6 ROIs per condition; two-way ANOVA). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001"
NCBI gene IDs:
"PINK1: 68943
Miro1: 59040"
Figure legend: "(I) Representative  confocal images of the  soma  of Miro1KO cortical neurons expressing Miro1WT and mutant forms of Miro1,   YFPParkin and MtDsRed without valinomycin treatment (Scale bars = 10 µm). Quantification of parkin colocalization with MtDsRed (YFPParkin signal overlapping MtDsRed  signal - intensity-adjusted) in the soma of Miro1KO and PINK1KO cortical neurons (n=12 cells all conditions per genotype over 3 neuronal preparations; one-way ANOVA). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001"
NCBI gene IDs:
"PINK1: 68943
Miro1: 59040"
Figure legend: "(J) Representative confocal images of the soma of  Miro1KO cortical neurons expressing WT and mutant forms of  Miro1, YFPParkin and  MtDsRed after 5 hours of valinomycin treatment (Scale bars = 10 µm). Quantification of  Parkin recruitment to mitochondria (Normalised to t=0) in  Miro1KO and PINK1KO cortical neurons following  valinomycin treatment (n=12 cells all conditions per genotype over 3 neuronal preparations, two-way ANOVA). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001 "
NCBI gene IDs:
"PINK1: 68943
Miro1: 59040"
Figure legend: "(A) Representative  blots of 4 and 12 months old hippocampal lysates from control, Miro1CKO and Miro2KO. B) Quantification of protein levels in 4 month lysates and 12 month  lysates (n=3 animals / genotype at 4 months and n=4 animals / genotype at 12 months; one-way ANOVA with post-hoc Dunnett's test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001."
NCBI gene IDs:
"Miro1: 59040
Miro2: 214952"
Figure legend: "C) Representative images of  hippocampal regions from control and Miro1CKO crossed with MitoDendra animals."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(D) Details from cortical  regions of control, Miro1CKO and Miro2KO animals at 12 months of age showing large mitochondrial  structures occurring only in the case of Miro1CKO animals."
NCBI gene IDs:
"Miro1: 59040
Miro2: 214952"
Figure legend: "(E) Electron microcopy images of control and  Miro1CKO brains showing the ultrastructure of the mitochondrial compartment. The mitochondrial units in Miro1CKO cells are enlarged and show altered cristae structure at 4 months of age. This process is progressive as mitochondrial  structures appear larger and with reduced intramitochondrial complexity at 12 months of  age."
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "Representative images (F) and quantification (G) of  Mfn1 and  Mfn2 staining in cortical  slices from 12 month WT and Miro1CKO mice  (n=4 animals / genotype; student's t-test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001. "
NCBI gene IDs:
"Miro1: 59040"
Figure legend: "(A) Representative  western blot images of  brain lysates from 12-month WT, Miro1CKO and Miro2KO mice immunoblotted  with the antibodies stated. (B) Quantification of  eIF2α and P-eIF2α band intensity (normalised to actin band  intensity) from 12-month WT, Miro1CKO and Miro2KO brain lysates. Phosphorylation of eIF2α  is defined as P-eIF2α/eIF2α (n=4 mice per genotype, unpaired t-test). Error bars represent s.e.m. Significance: *p&lt;0.05, **p&lt;0.01 and ***p&lt;0.001."
NCBI gene IDs:
"Miro1: 59040
Miro2: 214952"
Figure legend: "(C) Representative confocal images of cortical regions of 12-month mitoDendra-crossed WT and Miro1CKO mice stained with MAP2 and P-eIF2α. (D) Quantification of P-eIF2α signal intensity from cortical regions of 12-month  mitoDendra-crossed WT and Miro1CKO mice  (n=4 mice per genotype; unpaired t-test)."
NCBI gene IDs:
"Miro1: 59040"