Literature DB >> 16213388

Ophthalmologic findings in the Cornelia de Lange Syndrome.

Tamara Wygnanski-Jaffe1, John Shin, Enza Perruzza, Mohamed Abdolell, Laird G Jackson, Alex V Levin.   

Abstract

BACKGROUND: Cornelia de Lange Syndrome (CdLS) is a disorder caused in many patients by a mutation in the NIPBL gene with a dominant pattern of inheritance characterized by mental retardation, prenatal and postnatal growth retardation, upper-limb abnormalities, and characteristic facies. Few data exist concerning the ophthalmic findings in this syndrome.
METHODS: One hundred twenty individuals with CdLS underwent ophthalmic examination to ascertain the relative frequencies of oculofacial and ophthalmic abnormalities.
RESULTS: We confirmed the frequent findings of synophrys (99%), long lashes (99%), hypertrichosis of the brows (96%), ptosis (44%), epiphora (22%), nasolacrimal duct obstruction (16%), blepharitis (25%), and myopia (58%). In addition, we found peripapillary pigment (83%), and microcornea (21%), which have infrequently been mentioned in the literature.
CONCLUSION: Patients with CdLS can have multiple eye problems. Many of these problems can be readily treated, including myopia, blepharitis, nasolacrimal duct obstruction, and ptosis. Early examination is recommended for all children known or suspected to have CdLS.

Entities:  

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Year:  2005        PMID: 16213388     DOI: 10.1016/j.jaapos.2005.05.010

Source DB:  PubMed          Journal:  J AAPOS        ISSN: 1091-8531            Impact factor:   1.220


  8 in total

Review 1.  [Ophthalmological manifestations of Cornelia de Lange syndrome: Case report and review of the literature].

Authors:  G Avgitidou; C Cursiefen; L M Heindl
Journal:  Ophthalmologe       Date:  2015-05       Impact factor: 1.059

2.  Disruption of MBD5 contributes to a spectrum of psychopathology and neurodevelopmental abnormalities.

Authors:  J C Hodge; E Mitchell; V Pillalamarri; T L Toler; F Bartel; H M Kearney; Y S Zou; W H Tan; C Hanscom; S Kirmani; R R Hanson; S A Skinner; R C Rogers; D B Everman; E Boyd; C Tapp; S V Mullegama; D Keelean-Fuller; C M Powell; S H Elsea; C C Morton; J F Gusella; B DuPont; A Chaubey; A E Lin; M E Talkowski
Journal:  Mol Psychiatry       Date:  2013-04-16       Impact factor: 15.992

Review 3.  Cornelia de Lange syndrome, cohesin, and beyond.

Authors:  J Liu; I D Krantz
Journal:  Clin Genet       Date:  2009-10       Impact factor: 4.438

4.  Cornelia de Lange syndrome with optic disk pit: Novel association and review of literature.

Authors:  Bhamy Hariprasad Shenoy; Amit Gupta; Virender Sachdeva; Ramesh Kekunnaya
Journal:  Oman J Ophthalmol       Date:  2014-05

5.  Multiple organ system defects and transcriptional dysregulation in the Nipbl(+/-) mouse, a model of Cornelia de Lange Syndrome.

Authors:  Shimako Kawauchi; Anne L Calof; Rosaysela Santos; Martha E Lopez-Burks; Clint M Young; Michelle P Hoang; Abigail Chua; Taotao Lao; Mark S Lechner; Jeremy A Daniel; Andre Nussenzweig; Leonard Kitzes; Kyoko Yokomori; Benedikt Hallgrimsson; Arthur D Lander
Journal:  PLoS Genet       Date:  2009-09-18       Impact factor: 5.917

6.  Clinical and genetic study of 20 patients from China with Cornelia de Lange syndrome.

Authors:  Mingyan Hei; Xiangyu Gao; Lingqian Wu
Journal:  BMC Pediatr       Date:  2018-02-16       Impact factor: 2.125

7.  Surgical treatment of esotropia and unilateral ptosis in a patient with Cornelia de Lange syndrome.

Authors:  Won Jae Kim
Journal:  Yeungnam Univ J Med       Date:  2018-12-17

Review 8.  Diagnosis and management of Cornelia de Lange syndrome: first international consensus statement.

Authors:  Antonie D Kline; Joanna F Moss; Angelo Selicorni; Anne-Marie Bisgaard; Matthew A Deardorff; Peter M Gillett; Stacey L Ishman; Lynne M Kerr; Alex V Levin; Paul A Mulder; Feliciano J Ramos; Jolanta Wierzba; Paola Francesca Ajmone; David Axtell; Natalie Blagowidow; Anna Cereda; Antonella Costantino; Valerie Cormier-Daire; David FitzPatrick; Marco Grados; Laura Groves; Whitney Guthrie; Sylvia Huisman; Frank J Kaiser; Gerritjan Koekkoek; Mary Levis; Milena Mariani; Joseph P McCleery; Leonie A Menke; Amy Metrena; Julia O'Connor; Chris Oliver; Juan Pie; Sigrid Piening; Carol J Potter; Ana L Quaglio; Egbert Redeker; David Richman; Claudia Rigamonti; Angell Shi; Zeynep Tümer; Ingrid D C Van Balkom; Raoul C Hennekam
Journal:  Nat Rev Genet       Date:  2018-10       Impact factor: 53.242

  8 in total

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