Literature DB >> 27696664

A Say-Barber-Biesecker-Young-Simpson variant of Ohdo syndrome with a KAT6B 10-base pair palindromic duplication: A recurrent mutation causing a severe phenotype mixed with genitopatellar syndrome.

Yo Niida1, Yusuke Mitani2, Mondo Kuroda2, Ayano Yokoi2, Hiroyasu Nakagawa2, Akiko Kato2.   

Abstract

The Say-Barber-Biesecker-Young-Simpson variant of Ohdo syndrome (SBBYSS) (MIM# 603736) and genitopatellar syndrome (GPS) (MIM#606170) are allelic diseases caused by KAT6B mutation. Genotype-phenotype correlation is assumed, but a few patients manifest overlapping features of both syndromes. Here we report the case of a boy with SBBYSS. He had a KAT6B mutation previously reported in typical SBBYSS, but he also manifested severe developmental delay, as well as genital features and laryngomalacia requiring tracheostomy that conformed to GPS.
© 2016 Japanese Teratology Society.

Entities:  

Keywords:  Genitopatellar syndrome; Genotype-phenotype correlation; KAT6B; Say-Barber-Biesecker-Young-Simpson variant of Ohdo syndrome (SBBYSS)

Mesh:

Substances:

Year:  2017        PMID: 27696664     DOI: 10.1111/cga.12196

Source DB:  PubMed          Journal:  Congenit Anom (Kyoto)        ISSN: 0914-3505            Impact factor:   1.409


  2 in total

1.  Novel variants in KAT6B spectrum of disorders expand our knowledge of clinical manifestations and molecular mechanisms.

Authors:  Megan Yabumoto; Jessica Kianmahd; Meghna Singh; Maria F Palafox; Angela Wei; Kathryn Elliott; Dana H Goodloe; S Joy Dean; Catherine Gooch; Brianna K Murray; Erin Swartz; Samantha A Schrier Vergano; Meghan C Towne; Kimberly Nugent; Elizabeth R Roeder; Christina Kresge; Beth A Pletcher; Katheryn Grand; John M Graham; Ryan Gates; Natalia Gomez-Ospina; Subhadra Ramanathan; Robin Dawn Clark; Kimberly Glaser; Paul J Benke; Julie S Cohen; Ali Fatemi; Weiyi Mu; Kristin W Baranano; Jill A Madden; Cynthia S Gubbels; Timothy W Yu; Pankaj B Agrawal; Mary-Kathryn Chambers; Chanika Phornphutkul; John A Pugh; Kate A Tauber; Svetlana Azova; Jessica R Smith; Anne O'Donnell-Luria; Hannah Medsker; Siddharth Srivastava; Deborah Krakow; Daniela N Schweitzer; Valerie A Arboleda
Journal:  Mol Genet Genomic Med       Date:  2021-09-14       Impact factor: 2.183

2.  Genitopatellar syndrome: the first reported case in Japan.

Authors:  Satomi Okano; Akie Miyamoto; Ikue Fukuda; Hajime Tanaka; Kenichiro Hata; Tadashi Kaname; Yoichi Matsubara; Yoshio Makita
Journal:  Hum Genome Var       Date:  2018-05-28
  2 in total

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