The LisH Domain-Containing N-Terminal Fragment is Important for the Localization, Dimerization, and Stability of Katnal2 in Tetrahymena

Joachimiak, Ewa; Wacławek, Ewa; Niziolek, Michal; Osinka, Anna; Fabczak, Hanna; Gaertig, Jacek; Włoga, Dorota

doi:10.3390/cells9020292

Cited by 8 publications

(6 citation statements)

References 47 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…KATNAL2 is a member of the paralogue of katanin, which is a type of microtubule‐severing protein and critical regulator of microtubule dynamics in multiple species 30 . The generation of mature germ cells, including the sperm and oocyte, is tightly dependent on complex and precise regulation of meiosis in the primordial germ cells, in which microtubule severing and spindle assembly are indispensable 31–33 .…”

Section: Discussionmentioning

confidence: 99%

Biallelic mutations in KATNAL2 cause male infertility due to oligo‐astheno‐teratozoospermia

et al. 2021

View full text Add to dashboard Cite

Oligo‐astheno‐teratozoospermia (OAT) is a common cause of male infertility, and most of idiopathic OAT patients are thought to be caused by genetic defects. Here, we recruited 38 primary infertile patients with the OAT phenotype and 40 adult men with proven fertility for genetic analysis and identified biallelic mutations of KATNAL2 by whole‐exome sequencing in two cases. F013/II:1, from a consanguineous family, carried the KATNAL2 c.328C > T:p.Arg110X homozygous mutations. The other carried c.55A > G: p.Lys19Glu and c.169C > T: p Arg57Trp biallelic mutations. None of the KATNAL2 variants were found in the 40 adult men with proven fertility. The spermatozoa from patients with KATNAL2 biallelic mutations exhibited conspicuous defects in maturation, head morphology, and the structure of mitochondrial sheaths and flagella. KATNAL2 was mainly expressed in the pericentriolar material and mitochondrial sheath of the spermatozoa from control subjects, but it was undetectable in the spermatozoa from the patients. Furthermore, Katnal2 null male mice were infertile and displayed an OAT phenotype. Our results proved that the biallelic mutations in KATNAL2 cause male infertility and OAT in humans for the first time, to our knowledge, which could enrich the genetic defect spectrum of OAT and be beneficial for its accurate genetic screening and clinical diagnosis.

show abstract

Section: Discussionmentioning

confidence: 99%

Biallelic mutations in KATNAL2 cause male infertility due to oligo‐astheno‐teratozoospermia

et al. 2021

View full text Add to dashboard Cite

show abstract

“…Interestingly in KATNAL2, the MIT domain of KATNA1 and KATNAL1 is replaced by a lissencephaly homology (LisH) domain ( Figure 1 ; Cheung et al, 2016 ; Ververis et al, 2016 ). In the ciliate Tetrahymena thermophila , the LisH domain of the katanin homolog Kat2 is important for its stability, self-dimerization, and localization to the basal body and ciliary axoneme ( Joachimiak et al, 2020 ). Although the functional importance of the KATNAL2 LisH domain has yet to be determined in vertebrates, it is likely to regulate KATNAL2 localization, stabilization, self-association, and association with other proteins.…”

Section: Katanin Subunit Conservation and Domain Organizationmentioning

confidence: 99%

“…The function of katanin on the cilia has been widely studied using unicellular eukaryotes; for example, research investigating Tetrahymena and Chlamydomonas A- and B-subunit homologs has informed on katanin activity and localization on ciliary structures ( Waclawek et al, 2017 ; Joachimiak et al, 2020 ), as well as the biogenesis of motile cilia ( Sharma et al, 2007 ). Several recent studies have implicated katanin in the assembly, disassembly, and function of cilia in vertebrates ( Hu et al, 2014 ; Willsey et al, 2018 ; Mirvis et al, 2019 ).…”

Section: Katanin In Ciliary Homeostasis and Developmentmentioning

confidence: 99%

The Mammalian Family of Katanin Microtubule-Severing Enzymes

Lynn

Martinez

Nguyen

et al. 2021

Front. Cell Dev. Biol.

View full text Add to dashboard Cite

The katanin family of microtubule-severing enzymes is critical for cytoskeletal rearrangements that affect key cellular processes like division, migration, signaling, and homeostasis. In humans, aberrant expression, or dysfunction of the katanins, is linked to developmental, proliferative, and neurodegenerative disorders. Here, we review current knowledge on the mammalian family of katanins, including an overview of evolutionary conservation, functional domain organization, and the mechanisms that regulate katanin activity. We assess the function of katanins in dividing and non-dividing cells and how their dysregulation promotes impaired ciliary signaling and defects in developmental programs (corticogenesis, gametogenesis, and neurodevelopment) and contributes to neurodegeneration and cancer. We conclude with perspectives on future katanin research that will advance our understanding of this exciting and dynamic class of disease-associated enzymes.

show abstract

“…Structural analysis showed that HOS15 comprises four major motifs: the LisH motif, WD-40 domain, F-box-like motif, and DWD motif. The LisH motif is involved in protein dimerization (Choi et al, 2008;Joachimiak et al, 2020). The WD-40 motif is important for protein-protein interactions (Xu and Min, 2011).…”

Section: Introductionmentioning

confidence: 99%

Negative regulation of floral transition in Arabidopsis by HOS15-PWR-HDA9 complex

et al. 2023

View full text Add to dashboard Cite

Arabidopsis HOS15/PWR/HDA9 repressor complex, which is similar to the TBL1/NcoR1/HDAC complex in animals, plays a well-known role in epigenetic regulation. PWR and HDA9 have been reported to interact with each other and modulate the flowering time by repressing AGL19 expression, whereas HOS15 and HDA9, together with the photoperiodic evening complex, regulate flowering time through repression of GI transcription. However, the role of the HOS15/PWR/HDA9 core repressor complex as a functional unit in the regulation of flowering time is yet to be explored. In this study, we reported that the loss-of-function hos15-2/pwr/hda9 triple mutant accumulates higher transcript levels of AGL19 and exhibits an early flowering phenotype similar to those of hos15, pwr, and hda9 single mutants. Interestingly, the accumulation of HOS15 in the nucleus was drastically reduced in pwr and hda9 mutants. As a result, HOS15 could not perform its role in histone deacetylation or interaction with H3 in the nucleus. Furthermore, HOS15 is also associated with the same region of the AGL19 promoter known for PWR-HDA9 binding. The acetylation level of the AGL19 promoter was increased in the hos15-2 mutant, similar to the pwr and hda9 mutants. Therefore, our findings reveal that the HOS15/PWR/HDA9 repressor complex deacetylates the promoter region of AGL19, thereby negatively regulating AGL19 transcription, which leads to early flowering in Arabidopsis.

show abstract

The LisH Domain-Containing N-Terminal Fragment is Important for the Localization, Dimerization, and Stability of Katnal2 in Tetrahymena

Cited by 8 publications

References 47 publications

Biallelic mutations in KATNAL2 cause male infertility due to oligo‐astheno‐teratozoospermia

Biallelic mutations in KATNAL2 cause male infertility due to oligo‐astheno‐teratozoospermia

The Mammalian Family of Katanin Microtubule-Severing Enzymes

Negative regulation of floral transition in Arabidopsis by HOS15-PWR-HDA9 complex

Contact Info

Product

Resources

About