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Property "Has-20abstract" (as page type) with input value "-5B-5BImage:MITOEAGLE-2Dlogo.jpg-7Cleft-7C100px-7Clink=http:-2F-2Fwww.mitoglobal.org-2Findex.php-2FMITOEAGLE-7CCOST-20Action-20MitoEAGLE-5D-5D-0AMutations-20in-20Wfs1-20gene,-20which-20are-20responsible-20for-20synthesis-20of-20transmembrane-20endoplasmatic-20reticulum-20(ER)-20protein-20wolframin,-20cause-20a-20multi-2Dtargeting-20disease-20Wolfram-20syndrome-20(WS).-20The-20first-20symptom-20of-20the-20WS-20is-20diabetes-20mellitus,-20followed-20in-20most-20cases-20by-20optic-20atrophy,-20diabetes-20insipidus-20and-20deafness.-20Also,-20link-20between-20Wfs1-20deficiency-20and-20mitochondrial-20dysfunction-20have-20shown,-20causing-20neurological-20degeneration,-20ataxia-20and-20alteration-20in-20heart-20and-20skeletal-20muscle-20performance-20-5B1-5D.-20WS-20is-20accompanied-20with-20progressive-20loss-20of-20pancreatic-20β-2Dcells-20cell-20caused-20by-20alterations-20in-20cellular-20Ca-3Csup-3E2+-3C-2Fsup-3E-20signaling-20related-20to-20ER-20stress-20and-20unfolded-20protein-20responses-20-5B2-5D.-20The-20precise-20role-20of-20wolframin-20and-20pathophysiology-20at-20organism-20level-20is-20still-20poorly-20understood.-20We-20used-20Wfs1KO-20mice-20characterized-20with-20impaired-20glucose-20tolerance-20and-20an-20activated-20pathway-20characteristic-20for-20metabolic-20diseases-20-5B3-5D.-20-0A-0AAim-20of-20the-20study-20is-20to-20follow-20alterations-20in-20OXPHOS-20capacity-20and-20phosphotransfer-20networks-20caused-20by-20Wfs1-20deficiency-20in-20different-20muscle-20types:-20oxidative-20heart-20and-20soleus-20muscle-20and-20glycolytic-20-27-27m.-20rectus-20femoris-27-27-20and-20-27-27m.-20gastrocnemius-27-27-20white.-20-0A-0AOur-20results-20showed-20that-20all-20the-20studied-20muscles-20Wfs1KO-20mouse-20has-20a-20maximal-20of-20ADP-2Ddependent-20respiration-20rate-20with-20glutamate-20and-20malate-20lower-20than-20that-20of-20wild-2Dtype-20(WT)-20animals.-20However,-20when-20pyruvate-20and-20malate-20is-20used-20as-20substrate,-20no-20significant-20difference-20was-20detected.-20At-20the-20same-20time-20the-20leak-20state-20without-20adenylates-20is-20higher-20in-20Wfs1KO-20in-20most-20muscles-20with-20both-20complex-20I-20substrate-20combinations-20used.-20At-20the-20same-20time-20respiration-20rates-20with-20succinate-20were-20unaffected-20by-20Wfs1-20deficiency.-20-20It-20indicates-20to-20metabolic-20alterations-20in-20supporting-20of-20OXPHOS-20at-20the-20level-20of-20complex-20I.-0A-0AFacilitated-20energy-20transfer-20by-20creatine-20kinase-20(CK),-20adenylate-20kinase-20(AK)-20and-20other-20energy-20transport-20pathways-20is-20governed-20by-20the-20metabolic-20status-20of-20the-20cell-20-5B4-5D.-20In-20Wfs1KO-20mice-20heart-20muscle-20AK-20pathway-20was-20more-20active-20than-20WT,-20while-20the-20creatine-20activated-20respiration-20is-20lower-20than-20WT.-20-20On-20the-20contrary,-20in-20the-20glycolytic-20m.-20rectus-20femoris-20the-20activity-20of-20AK-20pathway-20shows-20a-20slight-20decrease-20in-20comparison-20to-20the-20control.-20Results-20in-20energy-20transfer-20pathways-20in-20the-20heart-20and-20skeletal-20muscles-20of-20Wfs1KO-20mice-20indicate-20shift-20a-20in-20the-20energy-20pathway-20preferences.-20In-20addition,-20Wfs1KO-20mice-20showed-20changes-20in-20the-20coupling-20between-20OXPHOS-20and-20glycolysis-20in-20oxidative-20cardiac-20and-20in-20glycolytic-20gastrocnemius-20white-20muscle.-20These-20changes-20indicate-20to-20compensatory-20mechanism-20in-20response-20to-20metabolic-20alterations." contains invalid characters or is incomplete and therefore can cause unexpected results during a query or annotation process.

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