Literature DB >> 16900932

Severe form of Cockayne syndrome with varying clinical presentation and no photosensitivity in a family.

Fatma Müjgan Sonmez1, Figen Celep, Sibel Aylin Ugur, Aslihan Tolun.   

Abstract

We report six patients with Cockayne syndrome type B without photosensitivity. The patients are from the same inbred family and exhibit variable clinical features. The main clinical manifestations were progressive encephalopathy including intracranial calcification and white-matter lesions, dwarfism without growth hormone deficiency, senile appearance, mental and motor retardation, atrophy of subcutaneous fat tissue, severe pectus carinatus, and spasticity. Clinical photosensitivity was not observed in any patient. Other clinical findings were cataract, pigmentary retinopathy, and peripheral neuropathy. The onset of the disease was between 3 and 6 months of age. Molecular genetic analyses in the family established linkage to ERCC6, the gene responsible for Cockayne syndrome type B, confirming the clinical diagnosis.

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Year:  2006        PMID: 16900932     DOI: 10.1177/08830738060210041601

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  5 in total

1.  Neuroimaging in Cockayne syndrome.

Authors:  M Koob; V Laugel; M Durand; H Fothergill; C Dalloz; F Sauvanaud; H Dollfus; I J Namer; J-L Dietemann
Journal:  AJNR Am J Neuroradiol       Date:  2010-06-03       Impact factor: 3.825

2.  ERCC6 dysfunction presenting as progressive neurological decline with brain hypomyelination.

Authors:  Laila Shehata; Dimitre R Simeonov; Anja Raams; Lynne Wolfe; Adeline Vanderver; Xueli Li; Yan Huang; Shannon Garner; Cornelius F Boerkoel; Audrey Thurm; Gail E Herman; Cynthia J Tifft; Miao He; Nicolaas G J Jaspers; William A Gahl
Journal:  Am J Med Genet A       Date:  2014-09-22       Impact factor: 2.802

3.  Ultraviolet-B induces ERCC6 repression in lens epithelium cells of age-related nuclear cataract through coordinated DNA hypermethylation and histone deacetylation.

Authors:  Yong Wang; Fei Li; Guowei Zhang; Lihua Kang; Huaijin Guan
Journal:  Clin Epigenetics       Date:  2016-05-26       Impact factor: 6.551

4.  Heterogeneous clinical features in Cockayne syndrome patients and siblings carrying the same CSA mutations.

Authors:  Asma Chikhaoui; Ichraf Kraoua; Nadège Calmels; Sami Bouchoucha; Cathy Obringer; Khouloud Zayoud; Benjamin Montagne; Ridha M'rad; Sonia Abdelhak; Vincent Laugel; Miria Ricchetti; Ilhem Turki; Houda Yacoub-Youssef
Journal:  Orphanet J Rare Dis       Date:  2022-03-05       Impact factor: 4.123

5.  Identification and Characterization of a Novel Recurrent ERCC6 Variant in Patients with a Severe Form of Cockayne Syndrome B.

Authors:  Khouloud Zayoud; Ichraf Kraoua; Asma Chikhaoui; Nadège Calmels; Sami Bouchoucha; Cathy Obringer; Clément Crochemore; Dorra Najjar; Sinda Zarrouk; Najoua Miladi; Vincent Laugel; Miria Ricchetti; Ilhem Turki; Houda Yacoub-Youssef
Journal:  Genes (Basel)       Date:  2021-11-29       Impact factor: 4.096

  5 in total

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