Literature DB >> 9843043

Developmental delay and growth failure caused by a peroxisomal disorder, dihydroxyacetonephosphate acyltransferase (DHAP-AT) deficiency.

E R Elias1, M Mobassaleh, A K Hajra, A B Moser.   

Abstract

We describe a 6 1/2-year-old-girl presenting with a unique phenotype and dihydroxyacetonephosphate acyltransferase (DHAP-AT) deficiency (1.6% of control activity in cultured fibroblasts), a peroxisomal enzyme deficiency which was reported previously to cause rhizomelic chondroplasia punctata (RCDP). Her phenotype is less severe than that seen in classical RCDP, and is notable for short stature, microcataracts, normal limbs, mild hypotonia, and severe mental retardation. Epiphyseal stippling is present. This patient illustrates the variability of peroxisomal disorders whereby a specific defect in peroxisomal plasmalogen synthesis may lead to several phenotypes. Her case also suggests that children presenting with deficient growth, developmental delay, and epiphyseal stippling should be screened carefully for peroxisomal disorders, with measurement of plasmalogens in addition to very long chain fatty acids.

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Year:  1998        PMID: 9843043

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


  6 in total

1.  Modulation of peroxisome proliferator-activated receptor-alpha activity by N-acetyl cysteine attenuates inhibition of oligodendrocyte development in lipopolysaccharide stimulated mixed glial cultures.

Authors:  Manjeet K Paintlia; Ajaib S Paintlia; Mushfiquddin Khan; Inderjit Singh; Avtar K Singh
Journal:  J Neurochem       Date:  2008-01-12       Impact factor: 5.372

2.  Lipopolysaccharide-induced peroxisomal dysfunction exacerbates cerebral white matter injury: attenuation by N-acetyl cysteine.

Authors:  Manjeet K Paintlia; Ajaib S Paintlia; Miguel A Contreras; Inderjit Singh; Avtar K Singh
Journal:  Exp Neurol       Date:  2007-12-23       Impact factor: 5.330

Review 3.  Glycerophosphate/Acylglycerophosphate acyltransferases.

Authors:  Atsushi Yamashita; Yasuhiro Hayashi; Naoki Matsumoto; Yoko Nemoto-Sasaki; Saori Oka; Takashi Tanikawa; Takayuki Sugiura
Journal:  Biology (Basel)       Date:  2014-11-19

4.  The Trypanosoma brucei dihydroxyacetonephosphate acyltransferase TbDAT is dispensable for normal growth but important for synthesis of ether glycerophospholipids.

Authors:  Rachel Zufferey; Karim Pirani; Melanie Cheung-See-Kit; Sungsu Lee; Tyler A Williams; Daniel G Chen; Md Faruk Hossain
Journal:  PLoS One       Date:  2017-07-17       Impact factor: 3.240

5.  Peroxisome biogenesis disorders.

Authors:  Catherine Argyriou; Maria Daniela D'Agostino; Nancy Braverman
Journal:  Transl Sci Rare Dis       Date:  2016-11-07

6.  Oral batyl alcohol supplementation rescues decreased cardiac conduction in ether phospholipid-deficient mice.

Authors:  Hannes Todt; Fabian Dorninger; Peter J Rothauer; Claus M Fischer; Michael Schranz; Britta Bruegger; Christian Lüchtenborg; Janine Ebner; Karlheinz Hilber; Xaver Koenig; Fatma A Erdem; Vaibhavkumar S Gawali; Johannes Berger
Journal:  J Inherit Metab Dis       Date:  2020-06-05       Impact factor: 4.750

  6 in total

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