Literature DB >> 18950397

Microphthalmia with linear skin defects: a case report and review.

Vishakha M Sharma1, Arlene M Ruiz de Luzuriaga, Darrel Waggoner, Mark Greenwald, Sarah L Stein.   

Abstract

Microphthalmia with linear skin defects syndrome is an X-linked dominant disorder characterized by microphthalmia and other ocular anomalies as well as linear, jagged skin defects typically involving the scalp, face, neck, and upper trunk. Other associated characteristics include short stature, developmental delay, congenital heart defects, diaphragmatic hernia, agenesis of the corpus callosum, anencephaly, hydrocephalus, and seizures. Microphthalmia with linear skin defects syndrome is now known to be associated with a deletion of the X chromosome at Xp22. This is an area that has been found to include the HCCS gene, which encodes a holocytochrome c-type synthase believed to be critical in the regulation of apoptosis. We present a patient with classic clinical and genetic findings of MLS syndrome and discuss the primary characteristics and management of this disorder.

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Mesh:

Year:  2008        PMID: 18950397     DOI: 10.1111/j.1525-1470.2008.00724.x

Source DB:  PubMed          Journal:  Pediatr Dermatol        ISSN: 0736-8046            Impact factor:   1.588


  11 in total

1.  Expanding the phenotypic spectrum of PORCN variants in two males with syndromic microphthalmia.

Authors:  Paul D Brady; Hilde Van Esch; Nathalie Fieremans; Guy Froyen; Anne Slavotinek; Jan Deprest; Koenraad Devriendt; Joris R Vermeesch
Journal:  Eur J Hum Genet       Date:  2014-07-16       Impact factor: 4.246

2.  Whole exome sequence analysis of Peters anomaly.

Authors:  Eric Weh; Linda M Reis; Hannah C Happ; Alex V Levin; Patricia G Wheeler; Karen L David; Erin Carney; Brad Angle; Natalie Hauser; Elena V Semina
Journal:  Hum Genet       Date:  2014-09-03       Impact factor: 4.132

Review 3.  Mitochondrial cytochrome c biogenesis: no longer an enigma.

Authors:  Shalon E Babbitt; Molly C Sutherland; Brian San Francisco; Deanna L Mendez; Robert G Kranz
Journal:  Trends Biochem Sci       Date:  2015-06-11       Impact factor: 13.807

4.  Microphthalmia, Linear Skin Defects, Callosal Agenesis, and Cleft Palate in a Patient with Deletion at Xp22.3p22.2.

Authors:  Siulan Vendramini-Pittoli; Rosana Maria Candido-Souza; Rodrigo Gonçalves Quiezi; Roseli Maria Zechi-Ceide; Nancy Mizue Kokitsu-Nakata; Fernanda Sarquis Jehee; Lucilene Arilho Ribeiro-Bicudo; David R FitzPatrick; Maria Leine Guion-Almeida; Antonio Richieri-Costa
Journal:  J Pediatr Genet       Date:  2020-01-03

5.  Mutations in NDUFB11, encoding a complex I component of the mitochondrial respiratory chain, cause microphthalmia with linear skin defects syndrome.

Authors:  Vanessa A van Rahden; Erika Fernandez-Vizarra; Malik Alawi; Kristina Brand; Florence Fellmann; Denise Horn; Massimo Zeviani; Kerstin Kutsche
Journal:  Am J Hum Genet       Date:  2015-03-12       Impact factor: 11.025

Review 6.  Linear Skin Defects with Multiple Congenital Anomalies (LSDMCA): An Unconventional Mitochondrial Disorder.

Authors:  Alessia Indrieri; Brunella Franco
Journal:  Genes (Basel)       Date:  2021-02-11       Impact factor: 4.096

7.  The impairment of HCCS leads to MLS syndrome by activating a non-canonical cell death pathway in the brain and eyes.

Authors:  Alessia Indrieri; Ivan Conte; Giancarlo Chesi; Alessia Romano; Jade Quartararo; Rosarita Tatè; Daniele Ghezzi; Massimo Zeviani; Paola Goffrini; Ileana Ferrero; Paola Bovolenta; Brunella Franco
Journal:  EMBO Mol Med       Date:  2013-01-22       Impact factor: 12.137

8.  Microphthalmia with Linear Skin Defects (MLS) associated with Autism Spectrum Disorder (ASD) in a patient with Familial 12.9Mb Terminal Xp deletion.

Authors:  Lucia Margari; Annalisa Colonna; Francesco Craig; Mattia Gentile; Giustina Giannella; Anna Linda Lamanna; Anna Rosi Legrottaglie
Journal:  BMC Pediatr       Date:  2014-09-02       Impact factor: 2.125

9.  Mutations in COX7B cause microphthalmia with linear skin lesions, an unconventional mitochondrial disease.

Authors:  Alessia Indrieri; Vanessa Alexandra van Rahden; Valeria Tiranti; Manuela Morleo; Daniela Iaconis; Roberta Tammaro; Ilaria D'Amato; Ivan Conte; Isabelle Maystadt; Stephanie Demuth; Alex Zvulunov; Kerstin Kutsche; Massimo Zeviani; Brunella Franco
Journal:  Am J Hum Genet       Date:  2012-11-02       Impact factor: 11.025

10.  Familial cases of a submicroscopic Xp22.2 deletion: genotype-phenotype correlation in microphthalmia with linear skin defects syndrome.

Authors:  Sarah Vergult; Bart Leroy; Ilse Claerhout; Björn Menten
Journal:  Mol Vis       Date:  2013-02-06       Impact factor: 2.367

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