Literature DB >> 1789285

Mucolipidosis type IV: clinical manifestations and natural history.

D Chitayat1, C M Meunier, K A Hodgkinson, K Silver, M Flanders, I J Anderson, J M Little, D A Whiteman, S Carpenter.   

Abstract

The clinical manifestations and psychomotor development of five patients with mucolipidosis IV (MLIV) from three Ashkenazi-Jewish families are reported. The presenting symptoms were hypotonia, developmental delay, corneal clouding, and puffy eyelids. Four of the patients had convergent strabismus and none progressed beyond a developmental age of 15 months. One patient died of aspiration at 17 years while the oldest patient entered puberty at 20 years, developed a coarse face at 30 years, and is now 32 years old. Histopathological studies in four patients showed storage changes characteristic of MLIV.

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Year:  1991        PMID: 1789285     DOI: 10.1002/ajmg.1320410310

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  22 in total

1.  G(M2)-ganglioside metabolism in situ in mucolipidosis IV fibroblasts.

Authors:  S Raghavan; E Leshinsky; E H Kolodny
Journal:  Neurochem Res       Date:  1999-04       Impact factor: 3.996

2.  Mucolipidosis type IV: abnormal transport of lipids to lysosomes.

Authors:  R Bargal; G Bach
Journal:  J Inherit Metab Dis       Date:  1997-09       Impact factor: 4.982

3.  Genetics of the human face: Identification of large-effect single gene variants.

Authors:  Daniel J M Crouch; Bruce Winney; Willem P Koppen; William J Christmas; Katarzyna Hutnik; Tammy Day; Devendra Meena; Abdelhamid Boumertit; Pirro Hysi; Ayrun Nessa; Tim D Spector; Josef Kittler; Walter F Bodmer
Journal:  Proc Natl Acad Sci U S A       Date:  2018-01-04       Impact factor: 11.205

4.  Novel degenerative and developmental defects in a zebrafish model of mucolipidosis type IV.

Authors:  Huiqing Li; Wuhong Pei; Sivia Vergarajauregui; Patricia M Zerfas; Nina Raben; Shawn M Burgess; Rosa Puertollano
Journal:  Hum Mol Genet       Date:  2017-07-15       Impact factor: 6.150

5.  Quantitative neuroimaging in mucolipidosis type IV.

Authors:  Raphael Schiffmann; Joan Mayfield; Caren Swift; Igor Nestrasil
Journal:  Mol Genet Metab       Date:  2013-11-21       Impact factor: 4.797

Review 6.  Mucolipidosis type IV: an update.

Authors:  Kazuyo Wakabayashi; Ann Marie Gustafson; Ellen Sidransky; Ehud Goldin
Journal:  Mol Genet Metab       Date:  2011-06-16       Impact factor: 4.797

7.  A TRP homolog in Saccharomyces cerevisiae forms an intracellular Ca(2+)-permeable channel in the yeast vacuolar membrane.

Authors:  C P Palmer; X L Zhou; J Lin; S H Loukin; C Kung; Y Saimi
Journal:  Proc Natl Acad Sci U S A       Date:  2001-06-26       Impact factor: 11.205

Review 8.  Mucolipin 1: endocytosis and cation channel--a review.

Authors:  Gideon Bach
Journal:  Pflugers Arch       Date:  2004-11-27       Impact factor: 3.657

Review 9.  Current concepts in the neuropathogenesis of mucolipidosis type IV.

Authors:  Lauren C Boudewyn; Steven U Walkley
Journal:  J Neurochem       Date:  2018-08-30       Impact factor: 5.372

10.  Fingolimod phosphate inhibits astrocyte inflammatory activity in mucolipidosis IV.

Authors:  Laura D Weinstock; Amanda M Furness; Shawn S Herron; Sierra S Smith; Sitara B Sankar; Samantha G DeRosa; Dadi Gao; Molly E Mepyans; Anna Scotto Rosato; Diego L Medina; Ayelet Vardi; Natalia S Ferreira; Soo Min Cho; Anthony H Futerman; Susan A Slaugenhaupt; Levi B Wood; Yulia Grishchuk
Journal:  Hum Mol Genet       Date:  2018-08-01       Impact factor: 6.150

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