Literature DB >> 29681106

Van Maldergem syndrome and Hennekam syndrome: Further delineation of allelic phenotypes.

Ivan Ivanovski1,2, Susan Akbaroghli3, Marzia Pollazzon1, Chiara Gelmini1, Stefano Giuseppe Caraffi1,4, Mahboubeh Mansouri5, Zahra Chavoshzadeh5, Simonetta Rosato1, Valeria Polizzi6, Giancarlo Gargano7, Marielle Alders8, Livia Garavelli1, Raoul C Hennekam9.   

Abstract

Biallelic variants in FAT4 are associated with the two disorders, Van Maldergem syndrome (VMS) (n = 11) and Hennekam syndrome (HS) (n= 40). Both conditions are characterized by a typical facial gestalt and mild to moderate intellectual disability, but differ in the occurrence of neonatal hypotonia and feeding problems, hearing loss, tracheal anomalies, and osteopenia in VMS, and lymphedema in HS. VMS can be caused by autosomal recessive variants in DCHS1 as well, and HS can also be caused by autosomal recessive variants in CCBE1 and ADAMTS3. Here we report two siblings with VMS and one girl with HS, all with FAT4 variants, and provide an overview of the clinical findings in all patients reported with FAT4 variants. Our comparison of the complete phenotypes of patients with VMS and HS indicates a resemblance of several signs, but differences in several other main signs and symptoms, each of marked importance for affected individuals.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  ADAMTS3; CCBE1; DCHS1; FAT4; Hennekam syndrome; Van Maldergem syndrome; lymphedema

Mesh:

Substances:

Year:  2018        PMID: 29681106     DOI: 10.1002/ajmg.a.38652

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


  5 in total

1.  ALPK1 hotspot mutation as a driver of human spiradenoma and spiradenocarcinoma.

Authors:  Mamunur Rashid; Michiel van der Horst; Thomas Mentzel; Francesca Butera; Ingrid Ferreira; Alena Pance; Arno Rütten; Bostjan Luzar; Zlatko Marusic; Nicolas de Saint Aubain; Jennifer S Ko; Steven D Billings; Sofia Chen; Marie Abi Daoud; James Hewinson; Sandra Louzada; Paul W Harms; Guia Cerretelli; Carla Daniela Robles-Espinoza; Rajiv M Patel; Louise van der Weyden; Chris Bakal; Jason L Hornick; Mark J Arends; Thomas Brenn; David J Adams
Journal:  Nat Commun       Date:  2019-05-17       Impact factor: 14.919

Review 2.  A multi-disciplinary, comprehensive approach to management of children with heterotaxy.

Authors:  Thomas G Saba; Gabrielle C Geddes; Stephanie M Ware; David N Schidlow; Pedro J Del Nido; Nathan S Rubalcava; Samir K Gadepalli; Terri Stillwell; Anne Griffiths; Laura M Bennett Murphy; Andrew T Barber; Margaret W Leigh; Necia Sabin; Adam J Shapiro
Journal:  Orphanet J Rare Dis       Date:  2022-09-09       Impact factor: 4.303

3.  FAT4 identified as a potential modifier of orofacial cleft laterality.

Authors:  Sarah W Curtis; Daniel Chang; Miranda R Sun; Michael P Epstein; Jeffrey C Murray; Eleanor Feingold; Terri H Beaty; Seth M Weinberg; Mary L Marazita; Robert J Lipinski; Jenna C Carlson; Elizabeth J Leslie
Journal:  Genet Epidemiol       Date:  2021-06-15       Impact factor: 2.135

4.  Primary intestinal lymphangiectasia diagnosed by video capsule endoscopy in a patient with immunodeficiency presenting with Morganella morganii bacteraemia.

Authors:  John M Cunningham; Sansrita Nepal; Aimee E Truesdale
Journal:  BMJ Case Rep       Date:  2020-09-13

5.  Requirement of FAT and DCHS protocadherins during hypothalamic-pituitary development.

Authors:  Emily J Lodge; Paraskevi Xekouki; Tatiane S Silva; Cristiane Kochi; Carlos A Longui; Fabio R Faucz; Alice Santambrogio; James L Mills; Nathan Pankratz; John Lane; Dominika Sosnowska; Tina Hodgson; Amanda L Patist; Philippa Francis-West; Francoise Helmbacher; Constantine Stratakis; Cynthia L Andoniadou
Journal:  JCI Insight       Date:  2020-10-27
  5 in total

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