| Molecular genetics, neuroimaging outcomes, and structural analyses of novel and recurrent variants of WDR62 gene in two consanguineous Pakistani families with autosomal recessive primary microcephaly. | Molecular genetics, neuroimaging outcomes, and structural analyses of novel and recurrent variants of WDR62 gene in two consanguineous Pakistani families with autosomal recessive primary microcephaly.
Aslam K, Saeed A, Saeed HI, Bashir R, Abid H, Akhtar R, Habib N, Khan R, Asif R, Rafiq S, Asif M, Makhdoom EUH, Hussain MS, Baig SM, Anjum I. | 07/16/2024 |
| WDR62 variants contribute to congenital heart disease by inhibiting cardiomyocyte proliferation. | WDR62 variants contribute to congenital heart disease by inhibiting cardiomyocyte proliferation.
Hao L, Ma J, Wu F, Ma X, Qian M, Sheng W, Yan T, Tang N, Jiang X, Zhang B, Xiao D, Qian Y, Zhang J, Jiang N, Zhou W, Chen W, Ma D, Huang G., Free PMC Article | 07/16/2022 |
| WD repeat domain 62 (WDR62) promotes resistance of colorectal cancer to oxaliplatin through modulating mitogen-activated protein kinase (MAPK) signaling. | WD repeat domain 62 (WDR62) promotes resistance of colorectal cancer to oxaliplatin through modulating mitogen-activated protein kinase (MAPK) signaling.
Cai J, Su L, Luo W., Free PMC Article | 07/9/2022 |
| Neurological outcome in WDR62 primary microcephaly. | Neurological outcome in WDR62 primary microcephaly.
Ruaud L, Drunat S, Elmaleh-Bergès M, Ernault A, Guilmin Crepon S, MCPH Consortium, El Ghouzzi V, Auvin S, Verloes A, Passemard S. | 06/25/2022 |
| Systematic Analysis of the Oncogenic Role of WDR62 in Human Tumors. | Systematic Analysis of the Oncogenic Role of WDR62 in Human Tumors.
Bu Y, Zhang L, Ma X, Wang R, Zhang X, Li J., Free PMC Article | 01/15/2022 |
| WDR62 regulates spindle dynamics as an adaptor protein between TPX2/Aurora A and katanin. | WDR62 regulates spindle dynamics as an adaptor protein between TPX2/Aurora A and katanin.
Huang J, Liang Z, Guan C, Hua S, Jiang K., Free PMC Article | 10/23/2021 |
| WDR62 localizes katanin at spindle poles to ensure synchronous chromosome segregation. | WDR62 localizes katanin at spindle poles to ensure synchronous chromosome segregation.
Guerreiro A, De Sousa F, Liaudet N, Ivanova D, Eskat A, Meraldi P., Free PMC Article | 10/23/2021 |
| An integrated functional and clinical genomics approach reveals genes driving aggressive metastatic prostate cancer. | An integrated functional and clinical genomics approach reveals genes driving aggressive metastatic prostate cancer.
Das R, Sjöström M, Shrestha R, Yogodzinski C, Egusa EA, Chesner LN, Chen WS, Chou J, Dang DK, Swinderman JT, Ge A, Hua JT, Kabir S, Quigley DA, Small EJ, Ashworth A, Feng FY, Gilbert LA., Free PMC Article | 08/28/2021 |
| Further Delineation of Phenotype and Genotype of Primary Microcephaly Syndrome with Cortical Malformations Associated with Mutations in the WDR62 Gene. | Further Delineation of Phenotype and Genotype of Primary Microcephaly Syndrome with Cortical Malformations Associated with Mutations in the WDR62 Gene.
Slezak R, Smigiel R, Obersztyn E, Pollak A, Dawidziuk M, Wiszniewski W, Bekiesinska-Figatowska M, Rydzanicz M, Ploski R, Gawlinski P., Free PMC Article | 08/21/2021 |
| relative WDR62 mRNA expression was not statistically different in differentiated thyroid carcinoma tissues and goiter tissues | Significance of CEP78 and WDR62 gene expressions in differentiated thyroid carcinoma: Possible predictors of lateral lymph node metastasis.
Hammad MO, Elabbasy LM, Abd Elghaffar MA, Zaki MMA, Bazeed FB, Zahran MA. | 12/7/2019 |
| The expression levels of miR223 were significantly decreased in clinical bladder cancer (BC) specimens. The restoration of miR223 expression significantly inhibited tumor aggressiveness and induced apoptosis in BC cells. Direct binding between oncogenic WDR62 and miR223 was confirmed by luciferase assay. The knockdown of WDR62 significantly inhibited tumor aggressiveness and induced the apoptosis of BC cells. | Tumor‑suppressive microRNA‑223 targets WDR62 directly in bladder cancer.
Sugita S, Yoshino H, Yonemori M, Miyamoto K, Matsushita R, Sakaguchi T, Itesako T, Tatarano S, Nakagawa M, Enokida H. | 08/31/2019 |
| Study in mutant mice and human cerebral organoids showed that WDR62 deletion resulted in a reduction in the size of mouse brains and organoids due to the disruption of neural progenitor cells. WDR62 interacts with and promotes CEP170 localization to the basal body of primary cilium, where CEP170 recruits KIF2A to disassemble cilium. | Modeling microcephaly with cerebral organoids reveals a WDR62-CEP170-KIF2A pathway promoting cilium disassembly in neural progenitors.
Zhang W, Yang SL, Yang M, Herrlinger S, Shao Q, Collar JL, Fierro E, Shi Y, Liu A, Lu H, Herring BE, Guo ML, Buch S, Zhao Z, Xu J, Lu Z, Chen JF., Free PMC Article | 07/13/2019 |
| Haplotype analysis showed genetic relatedness between the families of the patients. Our findings expand the spectrum of mutations randomly distributed in the WDR62 gene. A review is also provided of the brain malformations described in WDR62 mutations in association with congenital microcephaly | A novel WDR62 missense mutation in microcephaly with abnormal cortical architecture and review of the literature.
Zombor M, Kalmár T, Nagy N, Berényi M, Telcs B, Maróti Z, Brandau O, Sztriha L. | 06/23/2019 |
| WDR62 coordinates the TNFalpha receptor signaling pathway to JNK activation through association with multiple kinases and the adaptor protein TRAF2. | WDR62 mediates TNFα-dependent JNK activation via TRAF2-MLK3 axis.
Prinz E, Aviram S, Aronheim A., Free PMC Article | 05/11/2019 |
| Exome sequencing led to the rapid and cost-effective identification of a novel homozygous mutation in WDR62 gene. | A novel non sense mutation in WDR62 causes autosomal recessive primary microcephaly: a case report.
Cherkaoui Jaouad I, Zrhidri A, Jdioui W, Lyahyai J, Raymond L, Egéa G, Taoudi M, El Mouatassim S, Sefiani A., Free PMC Article | 04/20/2019 |
| Wdr62 is involved in meiotic initiation via activating JNK signaling, which displays a novel mechanism for regulating meiotic initiation, and mutation of WDR62 is one of the potential etiologies of premature ovarian insufficiency in humans. | Wdr62 is involved in female meiotic initiation via activating JNK signaling and associated with POI in humans.
Zhou Y, Qin Y, Qin Y, Xu B, Guo T, Ke H, Chen M, Zhang L, Han F, Li Y, Chen M, Behrens A, Wang Y, Xu Z, Chen ZJ, Gao F., Free PMC Article | 01/19/2019 |
| Our findings demonstrate critical and diverse functions of WDR62 in neocortical development and provide insight into the mechanisms by which its disruption leads to a plethora of structural abnormalities. | Disruptions in asymmetric centrosome inheritance and WDR62-Aurora kinase B interactions in primary microcephaly.
Sgourdou P, Mishra-Gorur K, Saotome I, Henagariu O, Tuysuz B, Campos C, Ishigame K, Giannikou K, Quon JL, Sestan N, Caglayan AO, Gunel M, Louvi A., Free PMC Article | 11/10/2018 |
| We report a clinical feature, electroclinical findings, and clinical course of a patient with a severe phenotype of MCPH2 including microcephaly, refractory infantile spasms and intellectual disability. We detected a new homozygous splicing variant c.3335+1G>C in the WD repeat domain 62 (WDR62) gene, and an additional new heterozygous missense mutation c.1706T>A of G protein-coupled receptor 56 (GPR56) gene | A novel mutation of WDR62 gene associated with severe phenotype including infantile spasm, microcephaly, and intellectual disability.
Nardello R, Fontana A, Antona V, Beninati A, Mangano GD, Stallone MC, Mangano S. | 07/14/2018 |
| Authors demonstrated that WDR62 is a PLK1 substrate that is phosphorylated at Ser 897, and that this phosphorylation at the spindle poles promotes astral microtubule assembly to stabilize spindle orientation. | PLK1-mediated phosphorylation of WDR62/MCPH2 ensures proper mitotic spindle orientation.
Miyamoto T, Akutsu SN, Fukumitsu A, Morino H, Masatsuna Y, Hosoba K, Kawakami H, Yamamoto T, Shimizu K, Ohashi H, Matsuura S. | 03/24/2018 |
| Case Report: WDR62 missence mutations associated with early onset acanthosis and hyperkeratosis in a patient with autosomal recessive microcephaly type 2. | Novel mutations c.28G>T (p.Ala10Ser) and c.189G>T (p.Glu63Asp) in WDR62 associated with early onset acanthosis and hyperkeratosis in a patient with autosomal recessive microcephaly type 2.
Banerjee S, Chen H, Huang H, Wu J, Yang Z, Deng W, Chen D, Deng J, Su Y, Li Y, Wu C, Wang Y, Zeng H, Wang Y, Li X., Free PMC Article | 02/24/2018 |
| WDR62-overexpressing lung cancer cells exhibited an increase in cell growth. Moreover, the concurrent overexpression of WDR62 and TPX2, a WDR62-interacting protein that is also overexpressed in lung adenocarcinoma, induced centrosome amplification in the lung cells. | WDR62 overexpression is associated with a poor prognosis in patients with lung adenocarcinoma.
Shinmura K, Kato H, Kawanishi Y, Igarashi H, Inoue Y, Yoshimura K, Nakamura S, Fujita H, Funai K, Tanahashi M, Niwa H, Ogawa H, Sugimura H. | 09/30/2017 |
| A novel WDR62 missense mutation causes primary microcephaly in a large consanguineous Saudi family | A novel WDR62 mutation causes primary microcephaly in a large consanguineous Saudi family.
Naseer MI, Rasool M, Sogaty S, Chaudhary RA, Mansour HM, Chaudhary AG, Abuzenadah AM, Al-Qahtani MH., Free PMC Article | 07/8/2017 |
| The results confirm that mutations in ASPM or WDR62 are the major cause of autosomal recessive primary microcephaly in the Pakistani population. | Molecular analysis of 23 Pakistani families with autosomal recessive primary microcephaly using targeted next-generation sequencing.
Wang R, Khan A, Han S, Zhang X. | 05/27/2017 |
| Data show that CUL4B variants are associated with a wide range of cerebral malformations and suggest an important role in brain through its interaction with WDR62, a protein in which variants were identified in patients with cerebral malformations. | Variants in CUL4B are associated with cerebral malformations.
Vulto-van Silfhout AT, Nakagawa T, Bahi-Buisson N, Haas SA, Hu H, Bienek M, Vissers LE, Gilissen C, Tzschach A, Busche A, Müsebeck J, Rump P, Mathijssen IB, Avela K, Somer M, Doagu F, Philips AK, Rauch A, Baumer A, Voesenek K, Poirier K, Vigneron J, Amram D, Odent S, Nawara M, Obersztyn E, Lenart J, Charzewska A, Lebrun N, Fischer U, Nillesen WM, Yntema HG, Järvelä I, Ropers HH, de Vries BB, Brunner HG, van Bokhoven H, Raymond FL, Willemsen MA, Chelly J, Xiong Y, Barkovich AJ, Kalscheuer VM, Kleefstra T, de Brouwer AP., Free PMC Article | 09/12/2015 |
| Genetic factors contribute to modify the severity of the WDR62 phenotype. | Severe presentation of WDR62 mutation: is there a role for modifying genetic factors?
Poulton CJ, Schot R, Seufert K, Lequin MH, Accogli A, Annunzio GD, Villard L, Philip N, de Coo R, Catsman-Berrevoets C, Grasshoff U, Kattentidt-Mouravieva A, Calf H, de Vreugt-Gronloh E, van Unen L, Verheijen FW, Galjart N, Morris-Rosendahl DJ, Mancini GM. | 04/25/2015 |