Literature DB >> 19760652

Case report supporting that the Barber-Say and ablepharon macrostomia syndromes could represent one disorder.

Jennifer Haensel1, Nicolai Kohlschmidt, Susanne Pitz, Annerose Keilmann, Martin Zenker, Reinhard Ullmann, Thomas Haaf, Oliver Bartsch.   

Abstract

We report on a 7-year-old girl with unequivocal features of Barber-Say syndrome (BSS): generalized hypertrichosis especially at the back, dry lax skin, macrostomia, thin lips, cup-shaped ears, bulbous nose, hypoplastic nipples, and abnormal external genitalia. She also demonstrated conductive hearing impairment and microblepharon. BSS has been reported with ectropion (not present in our patient), but ablepharon and microblepharon (i.e., absent or hypoplastic eyelids) have always been considered as hallmarks of ablepharon macrostomia syndrome (AMS). This is the first report of microblepharon in BSS. Other authors have discussed that BSS and AMS could possibly represent one syndrome, and our report supports this hypothesis.

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Year:  2009        PMID: 19760652     DOI: 10.1002/ajmg.a.32993

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


  5 in total

1.  Barber-Say Syndrome and Ablepharon-Macrostomia Syndrome: A Patient's View.

Authors:  Beatrice De Maria; Tresia de Jager; Caitlin Sarubbi; Oliver Bartsch; Alberto Bianchi; Francesco Brancati; Hon-Yin B Chung; Albert David; Ariana Kariminejad; Maura Foresti; Marina Gallottini; Bertrand Isidor; Shannon Marchegiani; Fabiana Martins; Laura Mazzanti; Nathalie Roche; Ankur Singh; Cathy Stevens; Kenichi Suga; Martin Zenker; Raoul C Hennekam
Journal:  Mol Syndromol       Date:  2017-04-27

2.  Recurrent Mutations in the Basic Domain of TWIST2 Cause Ablepharon Macrostomia and Barber-Say Syndromes.

Authors:  Shannon Marchegiani; Taylor Davis; Federico Tessadori; Gijs van Haaften; Francesco Brancati; Alexander Hoischen; Haigen Huang; Elise Valkanas; Barbara Pusey; Denny Schanze; Hanka Venselaar; Anneke T Vulto-van Silfhout; Lynne A Wolfe; Cynthia J Tifft; Patricia M Zerfas; Giovanna Zambruno; Ariana Kariminejad; Farahnaz Sabbagh-Kermani; Janice Lee; Maria G Tsokos; Chyi-Chia R Lee; Victor Ferraz; Eduarda Morgana da Silva; Cathy A Stevens; Nathalie Roche; Oliver Bartsch; Peter Farndon; Eva Bermejo-Sanchez; Brian P Brooks; Valerie Maduro; Bruno Dallapiccola; Feliciano J Ramos; Hon-Yin Brian Chung; Cédric Le Caignec; Fabiana Martins; Witold K Jacyk; Laura Mazzanti; Han G Brunner; Jeroen Bakkers; Shuo Lin; May Christine V Malicdan; Cornelius F Boerkoel; William A Gahl; Bert B A de Vries; Mieke M van Haelst; Martin Zenker; Thomas C Markello
Journal:  Am J Hum Genet       Date:  2015-06-25       Impact factor: 11.025

3.  A case of Barber-Say syndrome in a male Japanese newborn.

Authors:  Kenichi Suga; Miki Shono; Aya Goji; Sato Matsuura; Miki Inoue; Masami Kawahito; Michiyo Kinoshita; Misa Takeda; Kazuhiro Mori
Journal:  Clin Case Rep       Date:  2014-09-04

4.  Barber-say syndrome: a confirmed case of TWIST2 gene mutation.

Authors:  Mulakkan David Yohannan; Jennifer Hilgeman; Katlin Allsbrook
Journal:  Clin Case Rep       Date:  2017-06-02

5.  General anesthesia of a Japanese infant with Barber-Say syndrome: a case report.

Authors:  Eisuke Hamaguchi; Yasuo M Tsutsumi; Katsuyoshi Kume; Yoko Sakai; Nami Kakuta; Yuta Uemura; Shinji Kawahito; Katsuya Tanaka
Journal:  JA Clin Rep       Date:  2016-06-02
  5 in total

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