Literature DB >> 31710778

Wiedemann-Steiner syndrome in two patients from Portugal.

Ana Grangeia1,2, Miguel Leão1,3, Carla P Moura1,2,3.   

Abstract

Wiedemann-Steiner syndrome (WSS) is a rare genetic disorder characterized by growth retardation, facial dysmorphism, hypertrichosis cubiti and neurodevelopment delay. It is caused by pathogenic variants in the KMT2A gene. This report describes two unrelated Portuguese patients, age 11 and 17 years, with a phenotype concordant with WSS and clinical and molecular diagnosis of WSS by the identification of two novel frameshift variants in the KMT2A gene. This work also highlights the presence of certain clinical features in patients with growth retardation and development delay and should draw attention to the diagnosis of WSS, when hirsutism, particularly hypertrichosis cubiti is present.
© 2019 Wiley Periodicals, Inc.

Entities:  

Keywords:  zzm321990KMT2A gene; autosomal dominant inheritance; development delay; growth retardation

Mesh:

Substances:

Year:  2019        PMID: 31710778     DOI: 10.1002/ajmg.a.61407

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


  6 in total

1.  Wiedemann-Steiner Syndrome: A Rare Differential Diagnosis of Neurodevelopmental Delay and Dysmorphic Features.

Authors:  Kursat Bora Carman; Emre Kaplan; Cefa Nil Aslan; Sinem Kocagil; Oguz Cilinigr; Coskun Yarar
Journal:  J Pediatr Genet       Date:  2020-09-23

2.  Expanding the genotypic and phenotypic spectrum in a diverse cohort of 104 individuals with Wiedemann-Steiner syndrome.

Authors:  Sarah E Sheppard; Ian M Campbell; Margaret H Harr; Nina Gold; Dong Li; Hans T Bjornsson; Julie S Cohen; Jill A Fahrner; Ali Fatemi; Jacqueline R Harris; Catherine Nowak; Cathy A Stevens; Katheryn Grand; Margaret Au; John M Graham; Pedro A Sanchez-Lara; Miguel Del Campo; Marilyn C Jones; Omar Abdul-Rahman; Fowzan S Alkuraya; Jennifer A Bassetti; Katherine Bergstrom; Elizabeth Bhoj; Sarah Dugan; Julie D Kaplan; Nada Derar; Karen W Gripp; Natalie Hauser; A Micheil Innes; Beth Keena; Neslida Kodra; Rebecca Miller; Beverly Nelson; Malgorzata J Nowaczyk; Zuhair Rahbeeni; Shay Ben-Shachar; Joseph T Shieh; Anne Slavotinek; Andrew K Sobering; Mary-Alice Abbott; Dawn C Allain; Louise Amlie-Wolf; Ping Yee Billie Au; Emma Bedoukian; Geoffrey Beek; James Barry; Janet Berg; Jonathan A Bernstein; Cheryl Cytrynbaum; Brian Hon-Yin Chung; Sarah Donoghue; Naghmeh Dorrani; Alison Eaton; Josue A Flores-Daboub; Holly Dubbs; Carolyn A Felix; Chin-To Fong; Jasmine Lee Fong Fung; Balram Gangaram; Amy Goldstein; Rotem Greenberg; Thoa K Ha; Joseph Hersh; Kosuke Izumi; Staci Kallish; Elijah Kravets; Pui-Yan Kwok; Rebekah K Jobling; Amy E Knight Johnson; Jessica Kushner; Bo Hoon Lee; Brooke Levin; Kristin Lindstrom; Kandamurugu Manickam; Rebecca Mardach; Elizabeth McCormick; D Ross McLeod; Frank D Mentch; Kelly Minks; Colleen Muraresku; Stanley F Nelson; Patrizia Porazzi; Pavel N Pichurin; Nina N Powell-Hamilton; Zoe Powis; Alyssa Ritter; Caleb Rogers; Luis Rohena; Carey Ronspies; Audrey Schroeder; Zornitza Stark; Lois Starr; Joan Stoler; Pim Suwannarat; Milen Velinov; Rosanna Weksberg; Yael Wilnai; Neda Zadeh; Dina J Zand; Marni J Falk; Hakon Hakonarson; Elaine H Zackai; Fabiola Quintero-Rivera
Journal:  Am J Med Genet A       Date:  2021-03-30       Impact factor: 2.578

Review 3.  Chromatin Structure and Dynamics: Focus on Neuronal Differentiation and Pathological Implication.

Authors:  Sophie A Nothof; Frédérique Magdinier; Julien Van-Gils
Journal:  Genes (Basel)       Date:  2022-04-02       Impact factor: 4.141

4.  Trio-WES reveals a novel de novo missense mutation of KMT2A in a Chinese patient with Wiedemann-Steiner syndrome: A case report.

Authors:  Xiong Wang; Guijiao Zhang; Yanjun Lu; Xiaoping Luo; Wei Wu
Journal:  Mol Genet Genomic Med       Date:  2020-12-15       Impact factor: 2.183

5.  Three de novo variants in KMT2A (MLL) identified by whole exome sequencing in patients with Wiedemann-Steiner syndrome.

Authors:  Sukun Luo; Bo Bi; Wenqian Zhang; Rui Zhou; Wei Chen; Peiwei Zhao; Yufeng Huang; Li Yuan; Xuelian He
Journal:  Mol Genet Genomic Med       Date:  2021-09-01       Impact factor: 2.183

6.  Phenotypic Variation in Two Siblings Affected with Shwachman-Diamond Syndrome: The Use of Expert Variant Interpreter (eVai) Suggests Clinical Relevance of a Variant in the KMT2A Gene.

Authors:  Ibrahim Taha; Federica De Paoli; Selena Foroni; Susanna Zucca; Ivan Limongelli; Marco Cipolli; Cesare Danesino; Ugo Ramenghi; Antonella Minelli
Journal:  Genes (Basel)       Date:  2022-07-23       Impact factor: 4.141

  6 in total

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