Literature DB >> 8193482

Multisystem triglyceride storage disorder without ichthyosis in two siblings.

R Wessalowski1, H Schroten, E Neuen-Jacob, H Reichmann, B C Melnik, H G Lenard, T Voit.   

Abstract

A four-year-old boy presented with hepatomegaly, vacuolized granulocytes (Jordans' anomaly) and slightly progressive myopathy as signs of multisystem triglyceride storage disease. The nine-year-old sister of the patient also showed Jordans' anomaly and early fatigability, but no overt weakness. Biochemical analysis revealed normal values for carnitines, carnitine palmityl transferase in serum and striated muscle, and beta-oxidation enzymes in striated muscles. Distribution of non-membrane-bound lipids in granulocytes, fibroblasts, smooth muscle cells and striated muscle was compatible with Chanarin-Dorfman syndrome. In contrast to Chanarin-Dorfman syndrome, our patients lacked congenital ichthyosis.

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Year:  1994        PMID: 8193482     DOI: 10.1111/j.1651-2227.1994.tb12960.x

Source DB:  PubMed          Journal:  Acta Paediatr        ISSN: 0803-5253            Impact factor:   2.299


  3 in total

1.  Jordans' anomaly.

Authors:  K Rajeevan; K R Anandan; K P Vinayan; K E Urmila; K P Aravindan
Journal:  Indian J Pediatr       Date:  1999 Jul-Aug       Impact factor: 1.967

2.  Improved cytochemical method for detecting Jordans' bodies in neutral lipid storage diseases.

Authors:  Daniela Tavian; Roberto Colombo
Journal:  J Clin Pathol       Date:  2007-02-09       Impact factor: 3.411

3.  Contribution of novel ATGL missense mutations to the clinical phenotype of NLSD-M: a strikingly low amount of lipase activity may preserve cardiac function.

Authors:  Daniela Tavian; Sara Missaglia; Chiara Redaelli; Elena M Pennisi; Gloria Invernici; Ruediger Wessalowski; Robert Maiwald; Marcello Arca; Rosalind A Coleman
Journal:  Hum Mol Genet       Date:  2012-09-17       Impact factor: 6.150

  3 in total

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