Literature DB >> 26333423

A microdeletion encompassing PHF21A in an individual with global developmental delay and craniofacial anomalies.

Jonathan D J Labonne1,2, Julie Vogt3, Lisa Reali3, Il-Keun Kong4, Lawrence C Layman1,2,5, Hyung-Goo Kim1,2.   

Abstract

In Potocki-Shaffer syndrome (PSS), the full phenotypic spectrum is manifested when deletions are at least 2.1 Mb in size at 11p11.2. The PSS-associated genes EXT2 and ALX4, together with PHF21A, all map to this region flanked by markers D11S1393 and D11S1319. Being proximal to EXT2 and ALX4, a 1.1 Mb region containing 12 annotated genes had been identified by deletion mapping to explain PSS phenotypes except multiple exostoses and parietal foramina. Here, we report a male patient with partial PSS phenotypes including global developmental delay, craniofacial anomalies, minor limb anomalies, and micropenis. Using microarray, qPCR, RT-qPCR, and Western blot analyses, we refined the candidate gene region, which harbors five genes, by excluding two genes, SLC35C1 and CRY2, which resulted in a corroborating role of PHF21A in developmental delay and craniofacial anomalies. This microdeletion contains the least number of genes at 11p11.2 reported to date. Additionally, we also discuss the phenotypes observed in our patient with respect to those of published cases of microdeletions across the Potocki-Shaffer interval.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  11p11.2; ALX4; BHC80; BRAF35; CoREST; EXT2; GTF2I; H3K4me0; H3K4me1; H3K4me2; HDAC1; HDAC2; KDM1A; LSD1; MAPK8IP1; PHF21A; Potocki-Shaffer syndrome; ZMYM2; ZMYM3; ZNF217; craniofacial anomalies; developmental delay; haploinsufficiency; histone demethylase; histone eraser; histone reader; microdeletion; micropenis; positional cloning; repressor; tapering finger; unmethylated histone

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Substances:

Year:  2015        PMID: 26333423     DOI: 10.1002/ajmg.a.37344

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  8 in total

1.  Transcriptome Analysis Revealed Impaired cAMP Responsiveness in PHF21A-Deficient Human Cells.

Authors:  Robert S Porter; Yumie Murata-Nakamura; Hajime Nagasu; Hyung-Goo Kim; Shigeki Iwase
Journal:  Neuroscience       Date:  2017-05-29       Impact factor: 3.590

2.  Novel Pathogenic Variant (c.1171A>T) in PHF21A in a Female with Intellectual Disability and Craniofacial Anomalies.

Authors:  Cheonghwa Lee; Jung Yoon; Borae G Park; Baik-Lin Eun; Jung Ah Kwon
Journal:  Mol Syndromol       Date:  2022-03-09

3.  De novo truncating variants in PHF21A cause intellectual disability and craniofacial anomalies.

Authors:  Kohei Hamanaka; Yuji Sugawara; Takeyoshi Shimoji; Tone Irene Nordtveit; Mitsuhiro Kato; Mitsuko Nakashima; Hirotomo Saitsu; Toshimitsu Suzuki; Kazuhiro Yamakawa; Ingvild Aukrust; Gunnar Houge; Satomi Mitsuhashi; Atsushi Takata; Kazuhiro Iwama; Ahmed Alkanaq; Atsushi Fujita; Eri Imagawa; Takeshi Mizuguchi; Noriko Miyake; Satoko Miyatake; Naomichi Matsumoto
Journal:  Eur J Hum Genet       Date:  2018-11-28       Impact factor: 4.246

4.  Neurodevelopmental disease genes implicated by de novo mutation and copy number variation morbidity.

Authors:  Bradley P Coe; Holly A F Stessman; Arvis Sulovari; Madeleine R Geisheker; Trygve E Bakken; Allison M Lake; Joseph D Dougherty; Ed S Lein; Fereydoun Hormozdiari; Raphael A Bernier; Evan E Eichler
Journal:  Nat Genet       Date:  2018-12-17       Impact factor: 38.330

5.  Longitudinal assessment of neuronal 3D genomes in mouse prefrontal cortex.

Authors:  Amanda C Mitchell; Behnam Javidfar; Lucy K Bicks; Rachael Neve; Krassimira Garbett; Sharon S Lander; Karoly Mirnics; Hirofumi Morishita; Marcelo A Wood; Yan Jiang; Inna Gaisler-Salomon; Schahram Akbarian
Journal:  Nat Commun       Date:  2016-09-06       Impact factor: 14.919

Review 6.  Histone Lysine Methylation and Neurodevelopmental Disorders.

Authors:  Jeong-Hoon Kim; Jang Ho Lee; Im-Soon Lee; Sung Bae Lee; Kyoung Sang Cho
Journal:  Int J Mol Sci       Date:  2017-06-30       Impact factor: 5.923

7.  Disruption of PHF21A causes syndromic intellectual disability with craniofacial anomalies, epilepsy, hypotonia, and neurobehavioral problems including autism.

Authors:  Hyung-Goo Kim; Jill A Rosenfeld; Daryl A Scott; Gerard Bénédicte; Jonathan D Labonne; Jason Brown; Marianne McGuire; Sonal Mahida; Sakkubai Naidu; Jacqueline Gutierrez; Gaetan Lesca; Vincent des Portes; Ange-Line Bruel; Arthur Sorlin; Fan Xia; Yline Capri; Eric Muller; Dianalee McKnight; Erin Torti; Franz Rüschendorf; Oliver Hummel; Zeyaul Islam; Prasanna R Kolatkar; Lawrence C Layman; Duchwan Ryu; Il-Keun Kong; Suneeta Madan-Khetarpal; Cheol-Hee Kim
Journal:  Mol Autism       Date:  2019-10-22       Impact factor: 7.509

8.  11p11.12p12 duplication in a family with intellectual disability and craniofacial anomalies.

Authors:  Xuejiao Chen; Huihui Xu; Weiwu Shi; Feng Wang; Fenfen Xu; Yang Zhang; Jun Gan; Xiong Tian; Baojun Chen; Meizhen Dai
Journal:  BMC Med Genomics       Date:  2021-04-09       Impact factor: 3.063

  8 in total

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