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<lom:catalog>phaidra.vetmeduni.ac.at</lom:catalog>

  
<lom:entry>
  
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</lom:identifier>

  
<lom:identifier>
  
<lom:catalog>DOI</lom:catalog>

  
<lom:entry>
  
<lom:langstring xml:lang="x-none">10.26508/lsa.202101194</lom:langstring>

  
</lom:entry>

  
</lom:identifier>

  
<lom:title>
  
<lom:langstring xml:lang="en">The cation exchanger Letm1, circadian rhythms, and NAD(H) levels interconnect in diurnal zebrafish</lom:langstring>

  
</lom:title>

  
<lom:description>
  
<lom:langstring xml:lang="en">Mitochondria are fundamental for life and require balanced ion exchange to maintain proper functioning. The mitochondrial cation exchanger LETM1 sparks interest because of its pathophysiological role in seizures in the Wolf Hirschhorn Syndrome (WHS). Despite observation of sleep disorganization in epileptic WHS patients, and growing studies linking mitochondria and epilepsy to circadian rhythms, LETM1 has not been studied from the chronobiological perspective. Here we established a viable letm1 knock-out, using the diurnal vertebrate Danio rerio to study the metabolic and chronobiological consequences of letm1 deficiency. We report diurnal rhythms of Letm1 protein levels in wild-type fish. We show that mitochondrial nucleotide metabolism is deregulated in letm1-/- mutant fish, the rate-limiting enzyme of NAD+ production is up-regulated, while NAD+ and NADH pools are reduced. These changes were associated with increased expression amplitude of circadian core clock genes in letm1-/- compared with wild-type under light/dark conditions, suggesting decreased NAD(H) levels as a possible mechanism for circadian system perturbation in Letm1 deficiency. Replenishing NAD pool may ameliorate WHS-associated sleep and neurological disorders.</lom:langstring>

  
</lom:description>

  
<lom:language>eng</lom:language>

  
<lom:keyword>
  
<lom:langstring xml:lang="en">Critical Region; Hirschhorn; Metabolism; Protein; Gene</lom:langstring>

  
</lom:keyword>

  
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<lom:lifecycle>
  
<lom:datetime>2024-03-13T08:44:46.003Z</lom:datetime>

  
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<lom:langstring xml:lang="x-none">LOMv1.0</lom:langstring>

  
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<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Dao;Pauline;
FN:Pauline Dao
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Nowikovsky;Karin;
FN:Karin Nowikovsky
X-ORCID:https://orcid.org/0000-0001-8435-8410
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Tessmar-Raible;Kristin;
FN:Kristin Tessmar-Raible
X-ORCID:https://orcid.org/0000-0002-8038-1741
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Dinhopl;Nora;
FN:Nora Dinhopl
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Orel;Lukas;
FN:Lukas Orel
X-ORCID:https://orcid.org/0000-0001-6999-5833
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Hajny;Stefan;
FN:Stefan Hajny
X-ORCID:https://orcid.org/0000-0002-4178-7968
END:VCARD</lom:vcard>

  
</lom:centity>

  
<lom:centity>
  
<lom:vcard>BEGIN:VCARD
VERSION:3.0
N:Mekis;Ronald;
FN:Ronald Mekis
END:VCARD</lom:vcard>

  
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<lom:langstring xml:lang="de">Textdokument</lom:langstring>

  
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<lom:source>
  
<lom:langstring xml:lang="x-none">LOMv1.0</lom:langstring>

  
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<lom:langstring xml:lang="x-none">yes</lom:langstring>

  
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<lom:langstring xml:lang="x-t-cc-url">https://creativecommons.org/licenses/by/4.0</lom:langstring>

  
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<lom:technical>
  
<lom:format>application/pdf</lom:format>

  
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<lom:location>https://phaidra.vetmeduni.ac.at/o:2714</lom:location>

  
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<lom:source>
  
<lom:langstring xml:lang="x-none">LOMv1.0</lom:langstring>

  
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