Literature DB >> 10649795

Dandy-Walker anomaly in Meckel-Gruber syndrome.

P Cincinnati1, M E Neri, A Valentini.   

Abstract

We report a fetus affected by Meckel-Gruber syndrome whose phenotype was characterized by macrocephaly, frontal bossing, a saddle nose, marked micrognathia, a distended abdomen, omphalocele, post-axial polydactyly and talipes equinovarus. The main neuropathological finding at autopsy was in a very large cyst located in an abnormally wide posterior cranial fossa consistent with a Dandy-Walker anomaly. Intestinal malrotation, enlarged cystic dysplastic kidneys and hepatic portal fibrosis coexisted. The occurrence of a Dandy-Walker malformation in Meckel-Gruber syndrome confirms a disturbance in rhombencephalon development. Although uncommon, it should be included among the central nervous anomalies representative of the syndrome.

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Year:  2000        PMID: 10649795     DOI: 10.1097/00019605-200009010-00007

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  3 in total

1.  Molecular diagnostics of Meckel-Gruber syndrome highlights phenotypic differences between MKS1 and MKS3.

Authors:  Mark B Consugar; Vickie J Kubly; Donna J Lager; Cynthia J Hommerding; Wai Chong Wong; Egbert Bakker; Vincent H Gattone; Vicente E Torres; Martijn H Breuning; Peter C Harris
Journal:  Hum Genet       Date:  2007-03-22       Impact factor: 4.132

2.  Antenatal presentation of Bardet-Biedl syndrome may mimic Meckel syndrome.

Authors:  Houda Karmous-Benailly; Jelena Martinovic; Marie-Claire Gubler; Yoann Sirot; Laure Clech; Catherine Ozilou; Joëlle Auge; Nora Brahimi; Heather Etchevers; Eric Detrait; Chantal Esculpavit; Sophie Audollent; Géraldine Goudefroye; Marie Gonzales; Julia Tantau; Philippe Loget; Madeleine Joubert; Dominique Gaillard; Corinne Jeanne-Pasquier; Anne-Lise Delezoide; Marie-Odile Peter; Ghislaine Plessis; Brigitte Simon-Bouy; Hélène Dollfus; Martine Le Merrer; Arnold Munnich; Férechté Encha-Razavi; Michel Vekemans; Tania Attié-Bitach
Journal:  Am J Hum Genet       Date:  2005-01-21       Impact factor: 11.025

3.  MKS1 regulates ciliary INPP5E levels in Joubert syndrome.

Authors:  Gisela G Slaats; Christine R Isabella; Hester Y Kroes; Jennifer C Dempsey; Hendrik Gremmels; Glen R Monroe; Ian G Phelps; Karen J Duran; Jonathan Adkins; Sairam A Kumar; Dana M Knutzen; Nine V Knoers; Nancy J Mendelsohn; David Neubauer; Sotiria D Mastroyianni; Julie Vogt; Lisa Worgan; Natalya Karp; Sarah Bowdin; Ian A Glass; Melissa A Parisi; Edgar A Otto; Colin A Johnson; Friedhelm Hildebrandt; Gijs van Haaften; Rachel H Giles; Dan Doherty
Journal:  J Med Genet       Date:  2015-10-21       Impact factor: 6.318

  3 in total

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