Literature DB >> 18222743

Deletion 18q21.2q21.32 involving TCF4 in a boy diagnosed by CGH-array.

Joris Andrieux1, Frédéric Lepretre, Jean-Marie Cuisset, Alice Goldenberg, Bruno Delobel, Sylvie Manouvrier-Hanu, Muriel Holder-Espinasse.   

Abstract

We report on a 12 year-old boy presenting with severe developmental delay, dysmorphic features, limb anomalies, growth retardation, hypoplastic vermis and corpus callosum. Conventional and high-resolution cytogenetic analyses were normal. CGH-array allowed characterisation of a de novo 6.2 Mb 18q21.2q21.32 interstitial deletion involving TCF4, the recently identified gene responsible for Pitt-Hopkins syndrome (PHS). No tachypnoea-apnoea paroxysms were observed. We discuss the dysmorphic features particularly involving the ears, which might be helpful towards PHS and 18q21 deletion diagnosis.

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Year:  2008        PMID: 18222743     DOI: 10.1016/j.ejmg.2007.12.002

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  10 in total

Review 1.  Pitt-Hopkins Syndrome: A Review of Current Literature, Clinical Approach, and 23-Patient Case Series.

Authors:  Kimberly Goodspeed; Cassandra Newsom; Mary Ann Morris; Craig Powell; Patricia Evans; Sailaja Golla
Journal:  J Child Neurol       Date:  2018-01-10       Impact factor: 1.987

2.  Pitt-Hopkins syndrome: report of a case with a TCF4 gene mutation.

Authors:  Grazia Taddeucci; Alice Bonuccelli; Ilaria Mantellassi; Alessandro Orsini; Enrico Tarantino
Journal:  Ital J Pediatr       Date:  2010-02-02       Impact factor: 2.638

3.  The role of the TCF4 gene in the phenotype of individuals with 18q segmental deletions.

Authors:  Minire Hasi; Bridgette Soileau; Courtney Sebold; Annice Hill; Daniel E Hale; Louise O'Donnell; Jannine D Cody
Journal:  Hum Genet       Date:  2011-06-14       Impact factor: 4.132

4.  Neurologic and ocular phenotype in Pitt-Hopkins syndrome and a zebrafish model.

Authors:  Antje Brockschmidt; Alida Filippi; Peter Charbel Issa; Michael Nelles; Horst Urbach; Nicole Eter; Wolfgang Driever; Ruthild G Weber
Journal:  Hum Genet       Date:  2011-05-05       Impact factor: 4.132

5.  Pitt-Hopkins Syndrome.

Authors:  M Peippo; J Ignatius
Journal:  Mol Syndromol       Date:  2011-12-29

6.  MEF2C haploinsufficiency caused by either microdeletion of the 5q14.3 region or mutation is responsible for severe mental retardation with stereotypic movements, epilepsy and/or cerebral malformations.

Authors:  N Le Meur; M Holder-Espinasse; S Jaillard; A Goldenberg; S Joriot; P Amati-Bonneau; A Guichet; M Barth; A Charollais; H Journel; S Auvin; C Boucher; J-P Kerckaert; V David; S Manouvrier-Hanu; P Saugier-Veber; T Frébourg; C Dubourg; J Andrieux; D Bonneau
Journal:  J Med Genet       Date:  2009-07-09       Impact factor: 6.318

Review 7.  Pitt-Hopkins Syndrome and Differential Diagnosis: A Molecular and Clinical Challenge.

Authors:  Giuseppe Marangi; Marcella Zollino
Journal:  J Pediatr Genet       Date:  2015-09-25

8.  Identifying human disease genes through cross-species gene mapping of evolutionary conserved processes.

Authors:  Martin Poot; Alexandra Badea; Robert W Williams; Martien J Kas
Journal:  PLoS One       Date:  2011-05-04       Impact factor: 3.240

9.  Complex translocation disrupting TCF4 and altering TCF4 isoform expression segregates as mild autosomal dominant intellectual disability.

Authors:  Valerie Maduro; Barbara N Pusey; Praveen F Cherukuri; Paul Atkins; Christèle du Souich; Rosemarie Rupps; Marjolaine Limbos; David R Adams; Samarth S Bhatt; Patrice Eydoux; Amanda E Links; Anna Lehman; May C Malicdan; Christopher E Mason; Marie Morimoto; James C Mullikin; Andrew Sear; Clara Van Karnebeek; Pawel Stankiewicz; William A Gahl; Camilo Toro; Cornelius F Boerkoel
Journal:  Orphanet J Rare Dis       Date:  2016-05-14       Impact factor: 4.123

Review 10.  Pitt-Hopkins Syndrome: intellectual disability due to loss of TCF4-regulated gene transcription.

Authors:  J David Sweatt
Journal:  Exp Mol Med       Date:  2013-05-03       Impact factor: 8.718

  10 in total

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