Literature DB >> 24014347

An overview and online registry of microvillus inclusion disease patients and their MYO5B mutations.

K Joeri van der Velde1, Herschel S Dhekne, Morris A Swertz, Serena Sirigu, Virginie Ropars, Petra C Vinke, Trebor Rengaw, Peter C van den Akker, Edmond H H M Rings, Anne Houdusse, Sven C D van Ijzendoorn.   

Abstract

Microvillus inclusion disease (MVID) is one of the most severe congenital intestinal disorders and is characterized by neonatal secretory diarrhea and the inability to absorb nutrients from the intestinal lumen. MVID is associated with patient-, family-, and ancestry-unique mutations in the MYO5B gene, encoding the actin-based motor protein myosin Vb. Here, we review the MYO5B gene and all currently known MYO5B mutations and for the first time methodologically categorize these with regard to functional protein domains and recurrence in MYO7A associated with Usher syndrome and other myosins. We also review animal models for MVID and the latest data on functional studies related to the myosin Vb protein. To congregate existing and future information on MVID geno-/phenotypes and facilitate its quick and easy sharing among clinicians and researchers, we have constructed an online MOLGENIS-based international patient registry (www.MVID-central.org). This easily accessible database currently contains detailed information of 137 MVID patients together with reported clinical/phenotypic details and 41 unique MYO5B mutations, of which several unpublished. The future expansion and prospective nature of this registry is expected to improve disease diagnosis, prognosis, and genetic counseling.
© 2013 WILEY PERIODICALS, INC.

Entities:  

Keywords:  MYO5B; database; microvillus inclusion disease; myosin Vb; patient registry

Mesh:

Substances:

Year:  2013        PMID: 24014347     DOI: 10.1002/humu.22440

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  29 in total

Review 1.  Plasticity of the brush border - the yin and yang of intestinal homeostasis.

Authors:  Delphine Delacour; Julie Salomon; Sylvie Robine; Daniel Louvard
Journal:  Nat Rev Gastroenterol Hepatol       Date:  2016-02-03       Impact factor: 46.802

Review 2.  Trafficking Ion Transporters to the Apical Membrane of Polarized Intestinal Enterocytes.

Authors:  Amy Christine Engevik; James R Goldenring
Journal:  Cold Spring Harb Perspect Biol       Date:  2018-01-02       Impact factor: 10.005

Review 3.  Role of Polarity Proteins in the Generation and Organization of Apical Surface Protrusions.

Authors:  Gerard Apodaca
Journal:  Cold Spring Harb Perspect Biol       Date:  2018-01-02       Impact factor: 10.005

4.  Congenital Fatal Diarrhea in Newborns.

Authors:  Swapna Lingaldinna; Mangala Bharathi Sundaram; C N Kamalarathnam; Sumathi Bavanandam
Journal:  Indian J Pediatr       Date:  2017-08-26       Impact factor: 1.967

5.  An inducible mouse model for microvillus inclusion disease reveals a role for myosin Vb in apical and basolateral trafficking.

Authors:  Kerstin Schneeberger; Georg F Vogel; Hans Teunissen; Domenique D van Ommen; Harry Begthel; Layla El Bouazzaoui; Anke H M van Vugt; Jeffrey M Beekman; Judith Klumperman; Thomas Müller; Andreas Janecke; Patrick Gerner; Lukas A Huber; Michael W Hess; Hans Clevers; Johan H van Es; Edward E S Nieuwenhuis; Sabine Middendorp
Journal:  Proc Natl Acad Sci U S A       Date:  2015-09-21       Impact factor: 11.205

6.  Kinetic signatures of myosin-5B, the motor involved in microvillus inclusion disease.

Authors:  Sarah M Heissler; Krishna Chinthalapudi; James R Sellers
Journal:  J Biol Chem       Date:  2017-09-07       Impact factor: 5.157

Review 7.  Myosin-Driven Intracellular Transport.

Authors:  Margaret A Titus
Journal:  Cold Spring Harb Perspect Biol       Date:  2018-03-01       Impact factor: 10.005

8.  Disruption of Rab8a and Rab11a causes formation of basolateral microvilli in neonatal enteropathy.

Authors:  Qiang Feng; Edward M Bonder; Amy C Engevik; Lanjing Zhang; Matthew J Tyska; James R Goldenring; Nan Gao
Journal:  J Cell Sci       Date:  2017-06-08       Impact factor: 5.285

9.  Apical Membrane Alterations in Non-intestinal Organs in Microvillus Inclusion Disease.

Authors:  Cameron Schlegel; Victoria G Weis; Byron C Knowles; Lynne A Lapierre; Martin G Martin; Paul Dickman; James R Goldenring; Mitchell D Shub
Journal:  Dig Dis Sci       Date:  2017-12-07       Impact factor: 3.199

10.  Disrupted apical exocytosis of cargo vesicles causes enteropathy in FHL5 patients with Munc18-2 mutations.

Authors:  Georg F Vogel; Jorik M van Rijn; Iris M Krainer; Andreas R Janecke; Carsten Posovszky; Marta Cohen; Claire Searle; Prevost Jantchou; Johanna C Escher; Natalie Patey; Ernest Cutz; Thomas Müller; Sabine Middendorp; Michael W Hess; Lukas A Huber
Journal:  JCI Insight       Date:  2017-07-20
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