2019
DOI: 10.1172/jci98890
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Imbalanced mitochondrial function provokes heterotaxy via aberrant ciliogenesis

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Cited by 48 publications
(45 citation statements)
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“…Impairment of mitochondrial function results in longer cilia, while shorter cilia are generated when organelle activity is enhanced. Interestingly, he also found a significant reduction in mtDNA in a cohort of heterotaxy patients [160]. Some important reports have provided proof of an association between autophagy and cilia length; in detail, cilia elongation depends on mTOR pathway suppression and thus autophagy activation, which enables suppression of proteasomal degradation of the proteins that constitute cilia [162][163][164].…”
Section: Congenital Heart Diseasementioning
confidence: 95%
See 1 more Smart Citation
“…Impairment of mitochondrial function results in longer cilia, while shorter cilia are generated when organelle activity is enhanced. Interestingly, he also found a significant reduction in mtDNA in a cohort of heterotaxy patients [160]. Some important reports have provided proof of an association between autophagy and cilia length; in detail, cilia elongation depends on mTOR pathway suppression and thus autophagy activation, which enables suppression of proteasomal degradation of the proteins that constitute cilia [162][163][164].…”
Section: Congenital Heart Diseasementioning
confidence: 95%
“…Recently, some authors [160,161] have found that anomalies in cilia function are implicated in CHDs. Cilia are hair-like organelles that can be found on the surfaces of different types of cells.…”
Section: Congenital Heart Diseasementioning
confidence: 99%
“…The impairment of mitochondrial function has been involved in a variety of pathological conditions and diseases. For example, mitochondrial dysfunction could provoke heterotaxy via aberrant ciliogenesis and reduced the beating of cilia [29]. Our finding showed that the expression of mitochondrial biogenesis markers such as NRF1, NRF2, TFAM and COX4 were decreased in the epithelium of nasal polyp.…”
Section: Discussionmentioning
confidence: 58%
“…In addition to the number of mitochondria, the function of mitochondria is also critical for the beating of cilia [29]. Flow cytometry analysis revealed a depolarization of the mitochondrial membrane in Si-WDPCP HSECs and U0126-treated HSECs (Figure 5A-B).…”
Section: Wdpcp Stabilized Mitochondrial Function Through the Mapk/erkmentioning
confidence: 99%
“…In this issue of the JCI, Burkhalter et al (5) suggest a mechanism linking cilia and mitochondria in the development of multisystem heterotaxy syndromes. Using both pharmacological and genetic tools, they show that longer cilia are produced when mitochondrial function is impaired and shorter cilia are produced with enhanced mitochondrial function.…”
Section: Mitochondrial Dysfunction Cilial Length and Heterotaxymentioning
confidence: 99%